final case study submitted - sheffield
TRANSCRIPT
Integrated assessment of home based palliative
care with and without reinforced caregiver support:
‘A demonstration of INTEGRATE-HTA
methodological guidances’
The research leading to these results has received funding from the European Union Seventh Framework Programme ([FP7/2007-2013] [FP7/2007-2011]) under Grant
Agreement No. 306141.
The sole responsibility for the content of this publication lies with the authors. It does not necessarily reflect the opinion of the European Union. The European
Commission is not responsible for any use that may be made of the information contained therein.
Co-funded by the
European Union
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Foreword for the INTEGRATE-HTA Case Study
Professor Sheila Payne
You may have noticed that family members are central to all aspects of our lives, from
nurturing babies and young children to supporting frail older people. The availability of
family members is even more important during periods of sickness and disability. Evidence
suggests that they are crucial in supporting patients throughout advanced illness until death,
offering to facilitate preferences such as a home death but little is known about how best to
support them. Within Europe there are approximately 100 million family carers who
experience considerable physical, psychological, social and financial challenges, and
provide in total considerably more care than that delivered by health care workers within
national health care systems. According to NICE (2004), family carers refer to all people
with a close social and emotional bond, not just those related by kinship or marriage.
The case study focusing on home based palliative care, undertaken within the context of the
INTEGRATE-HTA project, is therefore greatly welcomed. The case study focuses on home
care, especially the additional support required by family carers to maintain a person with
advanced disease in their home (called 'reinforced'). This is extremely topical and relevant
to the work of clinicians based in primary care and also to hospital practitioners who need to
discharge patients nearing the end of life to the care of their family members. A key main
policy driver in the UK and many other European countries is to implement accessible and
timely home palliative and end of life care, to reduce the number of patients dying in
hospitals. This is based on the assumption that most patients prefer to die at home and that
hospital deaths are both more costly and potentially offer less quality on a number of
indicators. Sustaining the ability of family carers to continue to provide care at home is
therefore crucial. One of the major reasons for a break down in caregiving near the end of
life is excessive strain and burden upon family carers. The case study offers insights using
novel HTA methodology which will be relevant to all those planning, delivering and
developing new interventions to improve how we might help to prevent this caregiver
burden.
Professor Sheila Payne
Emeritus Professor International Observatory on End of Life Care Lancaster University, UK
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Executive Summary
What is already known about the topic
With changing disease patterns in Europe, increasingly complex health care technologies, such
as palliative care, have gained importance.
HTA has rarely been applied to highly complex health technologies. Indeed, contemporary
health technology assessment (HTA) is not adequately equipped to assess highly complex
technologies despite considerable progress in recent years.
What this case study report adds
This case study is designed to demonstrate the application of a number of the key concepts
and methods developed in the INTEGRATE-HTA project to home based palliative care with
and without an additional element of caregiver support as an example of a complex technology;
known as reinforced and non-reinforced home based palliative care respectively.
The INTEGRATE-HTA model, developed to enable integration of relevant assessment aspects,
is used to structure this report.
The case study reports on the application of some of the concepts and methods developed
within the INTEGRATE-HTA project to the assessment of effectiveness as well as economic,
sociocultural, ethical, and legal aspects; patient preferences and patient-specific moderators of
treatment and context and implementation issues related to reinforced and non-reinforced
home based palliative care.
The highly complex nature of reinforced and non-reinforced home based palliative care is
illustrated through an assessment of complexity characteristics.
The case study also included extensive lay and professional stakeholder involvement, using a
variety of methods, to inform the HTA process at key stages throughout the project.
Key messages are highlighted for both HTA research and for palliative care.
Implications for research and practice
The concepts and methods developed in the INTEGRATE-HTA project have been shown to be
feasible and to have the potential to offer added value but require further development and
application in the assessment of other complex technologies.
Reinforced and non-reinforced models palliative care are highly complex. The case study
findings offer some insights into their effectiveness as well as economic, sociocultural, ethical,
and legal issues; patient preferences and patient-specific moderators of treatment as well as
context and implementation issues.
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Contents
Foreword for the INTEGRATE-HTA Case Study .......................................................................................................2
Executive Summary ................................................................................................................................................3
Preface ....................................................................................................................................................................8
List of Tables ........................................................................................................................................................ 11
List of Figures ....................................................................................................................................................... 11
List of Boxes ......................................................................................................................................................... 11
Terminology used within this case study report ................................................................................................. 12
Glossary of Terms ................................................................................................................................................ 13
Abbreviations ....................................................................................................................................................... 14
Acknowledgements ............................................................................................................................................. 17
CHAPTER 1 ........................................................................................................................................................... 18
Background and Introduction to the project ............................................................................................... 18
Health Technology Assessment ................................................................................................................... 18
Complex health technologies ...................................................................................................................... 18
Aims of the INTEGRATE-HTA Project ........................................................................................................... 19
Design of the INTEGRATE-HTA Project ........................................................................................................ 19
Case Study on Palliative Care – A ‘Demonstration HTA’ .............................................................................. 19
Rationale for the selection of palliative care for the case study ................................................................. 20
Specialist palliative care as the focus for assessment ................................................................................. 20
Models of Palliative Care ............................................................................................................................. 21
Stakeholder involvement to determine the focus of the HTA..................................................................... 21
Non-reinforced models of home based palliative care (HBPC) ................................................................... 21
Reinforced models of home based palliative care (rHBPC) ......................................................................... 22
England as the context for the palliative care case study ........................................................................... 22
About this case study report ............................................................................................................................... 23
Purpose of the Case Study Report ............................................................................................................... 23
CHAPTER 2 ........................................................................................................................................................... 26
Steps 1 & 2 of the HTA model ..................................................................................................................... 26
The context for the HTA ............................................................................................................................... 28
Stakeholder Advisory Panels ........................................................................................................................ 30
Consultations with stakeholders in seven countries highlighted a range of similar issues. ........................ 31
Summary of Step 1 and Step 2 ..................................................................................................................... 35
References ................................................................................................................................................... 35
CHAPTER 3 ........................................................................................................................................................... 36
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Step 3 – Evidence Collection ........................................................................................................................ 36
Overview of Step 3 ....................................................................................................................................... 36
Background .................................................................................................................................................. 38
Aims ............................................................................................................................................................. 38
Methods ....................................................................................................................................................... 38
Findings ........................................................................................................................................................ 41
The economic aspects of home and reinforced home based palliative care ............................................. 54
Background .................................................................................................................................................. 54
Aims ............................................................................................................................................................. 54
Methods ....................................................................................................................................................... 54
Conclusions regarding economics ............................................................................................................... 78
Aim ............................................................................................................................................................... 81
Methods ....................................................................................................................................................... 81
Findings ........................................................................................................................................................ 82
Summary of findings .................................................................................................................................... 86
Reflections on the application of the guidance to assess ethical issues ..................................................... 86
Conclusion .................................................................................................................................................... 87
Socio-cultural aspects in (reinforced) home based palliative care ((r)HBPC) .............................................. 89
Background .................................................................................................................................................. 89
Aim ............................................................................................................................................................... 89
Methods ....................................................................................................................................................... 89
Reflections ................................................................................................................................................. 105
Conclusion .................................................................................................................................................. 109
Legal aspects of (reinforced) home based palliative care ......................................................................... 111
Background ................................................................................................................................................ 111
Aim ............................................................................................................................................................. 111
Methods ..................................................................................................................................................... 111
Findings ...................................................................................................................................................... 113
Conclusions ................................................................................................................................................ 114
Moderators of treatment outcome (reinforced home-based palliative care) .......................................... 114
Background ................................................................................................................................................ 114
Aim ............................................................................................................................................................. 114
Methods ..................................................................................................................................................... 114
Findings ...................................................................................................................................................... 115
Summary of findings from the literature ................................................................................................... 117
Patient preferences in (reinforced) home based palliative care (rHBPC).................................................. 119
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Background ................................................................................................................................................ 119
Aim ............................................................................................................................................................. 119
Methods ..................................................................................................................................................... 119
Findings ...................................................................................................................................................... 119
Summary of findings from the literature ................................................................................................... 120
Conclusions ................................................................................................................................................ 123
A qualitative review - contextual enablers and barriers to implementation ............................................ 124
Background ................................................................................................................................................ 124
Aim ............................................................................................................................................................. 124
Methods ..................................................................................................................................................... 125
Results ........................................................................................................................................................ 125
Summary of findings .................................................................................................................................. 125
Context ....................................................................................................................................................... 128
Conclusion .................................................................................................................................................. 136
Completing the evidence summary templates .......................................................................................... 139
Summary of Step 3 ..................................................................................................................................... 139
CHAPTER 4 ......................................................................................................................................................... 140
Step 4 – Mapping of the evidence ............................................................................................................. 140
Overview of Step 4 ..................................................................................................................................... 140
Assessment criteria .................................................................................................................................... 142
Results ........................................................................................................................................................ 142
Summary of Step 4 ..................................................................................................................................... 146
CHAPTER 5 ......................................................................................................................................................... 147
Overview of Step 5 ..................................................................................................................................... 147
Results ........................................................................................................................................................ 150
Step 1 Definition of the HTA objective and technology ............................................................................ 152
Step 2 Creation of a logic model to define evidence needs ...................................................................... 152
Step 3 Evidence collection and preparation .............................................................................................. 153
Step 4 Evidence processing and integration .............................................................................................. 154
Step 5 Decision-making.............................................................................................................................. 155
CHAPTER 6 ......................................................................................................................................................... 156
Discussion and Conclusions ....................................................................................................................... 156
Overview .................................................................................................................................................... 156
Lessons Learned ......................................................................................................................................... 156
The implications of the findings from the case study ................................................................................ 161
The feasibility and value of applying HTA approaches developed within the INTEGRATE-HTA project ... 161
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Key conclusions about reinforced and non-reinforced home based palliative care ................................. 164
Strengths of the case study ....................................................................................................................... 164
Limitations of the case study ..................................................................................................................... 165
Key areas for further research ................................................................................................................... 166
Recommendations ..................................................................................................................................... 168
Stakeholder Advisory Panel Members in England ..................................................................................... 174
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Preface
A ‘Demonstration HTA’
The prime purpose of the case study report is to demonstrate the application of the methods and
concepts for a more comprehensive, more patient–centred, and better integrated assessment –
hence this is a ‘demonstration HTA’, not a conventional HTA report. As such, the assessment draws
on a wide range of relevant evidence to test the new methods and concepts developed in the
INTEGRATE-HTA project.
We believe that this case study report may be of particular interest to those involved in Health
Technology Assessment (HTA) as it demonstrates the methods and concepts developed in the
INTEGRATE-HTA project. Additionally, the report may be of interest to the palliative care community
(i.e. those commissioning and delivering services) as it draws together a range of heterogenous
evidence that may assist decision making in a complex area of health care. The heterogenous
evidence presented about palliative care includes legal, ethical and sociocultural evidence, patient
preferences, moderators of treatment effect as well as effectiveness and cost effectiveness and
evidence about context and implementation.
Project Funding
The INTEGRATE-HTA project is co-funded by the European Union under the Seventh Framework
Programme (FP7-Health-2012-Innovation) under grant agreement n° 306141. This three year project
started on January 1st 2013 and ends December 31st 2015.
Disclaimer
The sole responsibility for the content of this presentation lies with the authors. It does not
necessarily reflect the opinion of the European Union. The European Commission is not responsible
for any use that may be made of the information contained therein.
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Authors of the case study report
Louise Brereton1, Philip Wahlster4, Kristin Bakke Lysdahl6, Kati Mozygemba4, Jake Burns8, James B.
Chilcott1, Sue Ward1, Jan Benedikt Brönneke4, Marcia Tummers7, Ralph van Hoorn7, Lisa
Pfadenhauer8, Stephanie Polus8, Christine Ingleton13, Clare Gardiner1,3, Gert Jan van der Wilt7,
Ansgar Gerhardus4, Anke Rohwer8,9, Eva Rehfuess8, Wija Oortwijn5, Pietro Refolo10, Dario
Sacchini10, Wojciech Leppert11, Aurelija Blazeviciene12, Antonio Gioacchino Spagnolo10, Louise
Preston1, Joe Clark1, Elizabeth Goyder1.
Contributors from the INTEGRATE-HTA team
Andrew Booth1, Bill Noble1,3, Jacqui Gath (Patient and Public Involvement (PPI) representative)1,
Suzy Paisley1, Anne Dehlfing4, Karen Hentschel4, Kim Weistra5, Bjørn Hofmann6, Wietske Kievit7,
Rob Baltussen7, Adriana Turriziani14.
On behalf of the INTEGRATE-HTA team.
Case study Administrator
Kate Chadwick13
Affiliations
1) ScHARR, The University of Sheffield, Sheffield, England
2) School of Nursing & Midwifery, The University of Sheffield, Sheffield, England
3) School of Nursing, University of Auckland, New Zealand
4) Department of Health Services Research, Health Sciences Bremen, University of Bremen,
Bremen, Germany. (Lead partner)
5) Health Unit, ECORYS Nederland B.V. Rotterdam, The Netherlands
6) Institute for Health and Society, University of Oslo, Oslo, Norway
7) Department for Health Evidence, Radboud University Medical Centre, Nijmegen, The
Netherlands
8) Institute for Medical Informatics, Biometry & Epidemiology, Ludwig-Maximilians-University
Munich, Germany
9) Centre for Evidence-based Health Care, Faculty of Medicine and Health Sciences,
Stellenbosch University, Stellenbosch, South Africa
10) Institute of Bioethics, “A.Gemelli” School of Medicine, Università Cattolica del Sacro Cuore,
Rome, Italy
11) Department of Palliative Medicine, Poznan University of Medical Sciences, Poznan, Poland
12) Department of Nursing and Care, Hospital of Lithuanian University of Health Sciences,
Kaunas Lithuania
13) School of Nursing and Midwifery, The University of Sheffield, Sheffield, England
14) Palliative care unit, Hospice “Villa Speranza”, Rome, Italy
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Correspondence
For all enquiries, please contact [email protected] or [email protected] or
Declare competing interests of authors
None
This report should be referenced as follows
Brereton, L., Wahlster, P., Lysdahl, K.B., Mozygemba, K., Burns, J., Chilcott, J.B., Ward, S.,
Brönneke, J.B., Tummers, M., Van Hoorn, R., Pfadenhauer, L., Polus, S., Ingleton, C., Gardiner, C.,
Van Der Wilt, G.J., Gerhardus, A., Rohwer, A., Rehfuess, E., Oortwijn, W., Refolo, P., Sacchini, D.,
Leppert, W., Blazeviciene, A., Spagnolo A.G., Preston, L., Clark, J., Goyder, E., On behalf of the
INTEGRATE-HTA team (2016) Integrated assessment of home based palliative care with and
without reinforced caregiver support: ‘A demonstration of INTEGRATE-HTA methodological
guidances’ [Online]. Available from: http://www.integrate-hta.eu/downloads or
http://www.sheffield.ac.uk/scharr/sections/ph/research/knowledgeintoaction
Authorship of the report
The report content reflects the depth and breadth of work undertaken for the INTEGRATE-HTA
project by a wide range of different teams across Europe. The report authorship reflects this as the
authors represent a range of expertise and are drawn from different professional and academic
backgrounds, from seven European countries. We believe that the diversity of backgrounds and
perspectives enhance the depth of coverage. This is reflected in the presentation of different aspects
and elements of HTA within this report.
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List of Tables
Table 3.1 Outcomes found in included studies .................................................................................................. 39
Table 3.2 Description of included reinforced home based palliative care interventions.................................. 43
Table 3.3 Overview of the discussions within the gap analysis and expert consultations ................................ 47
Table 3.4 Risk of bias in each study using Cochrane EPOC risk of bias tool ...................................................... 50
Table 3.5 Aspects of complexity relevant to considerations of the economics of rhbc ................................... 64
Table 3.6 Final scope for economic evaluation ................................................................................................ 66
Table 3.7 Elicitation parameter estimates and unit costs ................................................................................ 69
Table 3.8 Summary of changes in costs associated with reinforced carer support .......................................... 72
Table 3.9 Health and social care resource implications .................................................................................... 74
Table 3.10 Budget Impact .................................................................................................................................. 74
List of Figures
Figure 1.1 The INTEGRATE-HTA model for the integrated assessment of complex technologies .................... 24
Figure 2.1 Step 1 & 2 of the INTEGRATE-HTA model ......................................................................................... 27
Figure 2.2 The Logic Model Template …………………………………………………………………………………………………………..30
Figure 2.3 Logic model of reinforced and non-reinforced home-based palliative care .................................... 34
Figure 3.1 Step 3 of the INTEGRATE-HTA Model ............................................................................................... 37
Figure 3.2 Harvest plots showing intervention effects for patient outcomes ................................................... 45
Figure 3.3 Harvest plots showing intervention effects for informal caregiver outcomes ................................ 45
Figure 3.4 Reinforced home palliative care intervention theory ...................................................................... 62
Figure 3.5 Framework for ethical analysis in HTA of complex interventions .................................................... 82
Figure 3.6 Assessment process for the assessment of socio-cultural aspects .................................................. 90
Figure 3.7 Overview of the socio-cultural aspects represented in the socio-cultural framework .................... 91
Figure 3.8 Domains, Categories and themes of the context dimension of the CICI framework ..................... 128
Figure 3.9 Domains, Categories and themes of the implementation dimension of the CICI framework ....... 131
Figure 4.1 Step 4 of the INTEGRATE-HTA Model ............................................................................................. 141
Figure 4.2 Structure of HTA research question taking the HTA results into account .............................................
Figure 4.3 The extended logic model............................................................................................................... 145
Figure 5.1 Step 5 of the INTEGRATE-HTA Model ............................................................................................. 148
Figure 5.2 Mean weights of main assessment criteria in percent. .................................................................. 150
Figure 5.3 Relative weights for effectiveness for caregiver versus effectiveness for patients. ...................... 151
List of Boxes
Box 1.1 Aspects of complexity ........................................................................................................................... 19
Box 3.1 Legal issues of importance for different technologies ....................................................................... 112
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Terminology used within this case study report
There has been recognition of the urgent need to define and clarify the terminology used within
palliative care to enhance consistency in clinical practice and ensure that information is accessible to
a wide audience (Preston et al 2012, Gott et al 2013). Terminology differs across countries with
regard to both HTA and palliative care. A glossary of HTA terms can be found on the International
Network of Agencies for Health Technology Assessment (INAHTA) webpages (see Appendix 2 and
http://htaglossary.net/HomePage). A glossary of palliative care terminology is included at the outset
of this report and terminology is in Appendix 3.
Please note that the term informal caregiver – also known as a lay or ‘family’ caregiver is a contested
area, but the terms ‘informal caregiver’ or carer will be used. An informal caregiver is defined here as
an ‘unpaid family member, friend or neighbour’ who assumes the main lay responsibility for providing
assistance or care to the person anticipated to be nearing the end of their life (Stajduhar et al 2010).
Hence there is no requirement for the caregiver to have kinship ties to the care-recipient and their
role co-exists alongside that of professional services.
Applying the definition of technology given by HTAi (see above) interventions as understood by the
MRC are part of technologies so that in this proposal we will make no difference between
interventions and technologies. We acknowledge that palliative care does not consist of a single
intervention or technology and that this is not a new service. However, palliative care is highly
complex and therefore met the requirements for the case study in which we apply some of the new
concepts and methods for HTA developed within the INTEGRATE-HTA project.
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Glossary of Terms
Health technologies, according to a definition of the international society for health technology
assessment (HTAi), include pharmaceuticals, devices, diagnostics and treatments, and other clinical,
public health, and organisational interventions (HTAi n.d.). Interventions as understood by the MRC
(2008) are part of technologies. Hence we will make no difference between interventions and
technologies in this report.
Examples of terms
Palliative Care
provides relief from pain and other distressing symptoms;
affirms life and regards dying as a normal process;
intends neither to hasten or postpone death;
integrates the psychological and spiritual aspects of patient care;
offers a support system to help patients live as actively as possible until death;
offers a support system to help the family cope during the patients illness and in their own bereavement
Health Technology
Any intervention that may be used to promote health, prevent, diagnose or treat disease, or for
rehabilitation or long-term care
Health Technology Assessment
It may address the direct, intended consequences of technologies as well as their indirect,
unintended consequences. Its main purpose is to inform technology-related policymaking in health
care. HTA is conducted by interdisciplinary groups using explicit analytical frameworks drawing from
a variety of methods.
Intervention Design
Intervention design describes the “What?” of the intervention under the headings components and
execution. The components of the intervention can be categorised as technology and infrastructure;
education; or policy and regulations. The execution of the intervention comprises a more detailed
“prescription” of the intervention – timing (when), duration (how long), dose (how much) and intensity
(how often).
Intervention Delivery
“How?”, “Who?” and “Where?” of the intervention and distinguishes between delivery mechanisms, delivery agents and setting
References
Craig P, Dieppe P, Macintyre S et al (2008) Developing and evaluating complex interventions: New guidance 2008. Medical Research Council [on-line]. Available at http://www.mrc.ac.uk/complexinterventionsguicance.
Gott M, Ingleton C, Gardiner C et al (2013) Exploring the transition from curative care to palliative care: a systematic review of the literature (phase 1).in Transitions to palliative care for older people in acute hospitals: a mixed methods study Health Services and Delivery Research 1(1)1 Available at http://www.journalslibrary.nihr.ac.uk/__data/assets/pdf_file/0011/94277/FullReport-hsdr01110.pdf
Preston N, Short V, Hollingworth W et al (2012) MORECare research methods guidance development: recommendations for health economic evaluations in palliative and end of life care research. Palliative Medicine, 26(4): 541.
Stajduhar K, Funk L, Toye C et al (2010) Part 1: Home-based family caregiving at the end of life: a comprehensive review of published quantitative research (1998-2008), Palliative Medicine, 24: 573-93.
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Abbreviations
This list covers widely used abbreviations and acronyms, others are explained in the text or in notes
at the end of chapters.
ACD Appraisal Consultation Document
ACP Advance Care Plan
AHP Allied Health Professionals
ALS Amyotrophic lateral sclerosis
AMSTAR A Measurement Tool to Assess Systematic Reviews
AMKPS Australian-modified Karnofsky Performance Status
BMA British Medical Association
CBA Controlled Before & After Studies
CCG Clinical Commissioning Groups
CCT Controlled Clinical Trials
Cf E.g. (for example)
CHF Chronic Heart Failure
CME Continuing Medical Education
CICI Context and implementation for complex interventions
COPD Chronic Obstructive Pulmonary Disease
COPE Committee on Publications Ethics
DH Department of Health
DN District Nurse
EAPC European Association of Palliative Care
ED Emergency Department
EOL End of Life
ERG Evidence Review Group
EU European Union
EVIDEM Evidence and Value: Impact on Decision Making
FAD Final Appraisal Determination
GMC General Medical Council
GP General Practitioner
GPC Generalist Palliative Care
GSF Gold Standards Framework
HAH Hospice at Home
HBPC Home Based Palliative Care
HF Hearth Failure
HIV Human immunodeficiency virus
HRQoL Heart Related Quality of Life
HTA Health Technology Assessment
ICESCR International Covenant on Economic, Social and Cultural Rights
ICP Integrated Care Pathway
IPCOS Integrated Palliative Care Outcome Scale
INAHTA International Network of Agencies for Health Technology Assessment
IO International Organisations
ITSS Interrupted Time Series Studies
15
LA
Local Authority
LCP Liverpool Care Pathway
MCCC Marie Curie Cancer Centre
MCDA Multiple Criteria Decision Analysis
MCDA Multi-criteria decision analysis
MCNS Marie Curie Nursing Service
MDT Multi-Disciplinary Team
MORECare Methods Of Researching End of life Care
MND Motor Neurone Disease
MRC Medical Research Council
MTA Multiple technology appraisal
MTA Multiple Technology Appraisal
NAO National Audit Office
NCPC National Council for Palliative Care
NHS National Health Service
EOLCP National Health Service End of Life Care Programme
NICE National Institute for Clinical Excellence
NIHR National Institute for Health Research
NRES National Research Ethics Service
NTCRP North Trent Cancer Research Panel
OACC Outcome Assessment and Complexity Collaborative
OT Occupational Therapy
PC Palliative Care
PCSAG Palliative Care Studies Advisory Group
PCT Palliative Care Team
PCTs Primary Care Trusts
PHAC Public Health Advisory Committees
PHCT Palliative Home Care Teams
PICO Population, Intervention, Comparator and Outcome
PPI Patient & Public Involvement
PRISMA Preferred Reporting Items for Systematic Reviews and Meta-Analyses
Reflecting the Positive diveRsities of European PrIorities for reSearch &
Measurement in End-of-life cAre
PROMS Patient Reported Outcome Measures
PSS Personal Social Services
QALYS Quality Adjusted Life Years
QOL Quality of Life
RCGP Royal College of General Practitioners
RCT Randomised Controlled Trial
rHBPC Reinforced Home Based Palliative Care
RIFM Reconstructing Interpretative Frames Method
SAP Stakeholder Advisory Panel
SCM Supportive Care Measure
SD Standard Deviation
SPC Specialist Palliative Care
STA Single Technology Appraisal
16
STH Sheffield Teaching Hospitals
TAC Public Health Advisory Committees
TAR Technology Assessment Report
TIDier Template for Intervention Description and Replication
UK United Kingdom
UN United Nations
USA United States of America
VOC Views of Care
WHO World Health Organisation
WP Work Package
17
Acknowledgements
We would like to thank everyone who has contributed to the INTEGRATE-HTA project and the case
study report. Whilst it is possible to openly acknowledge the contribution of some individuals who
have agreed to this, many others cannot be named within this report as a result of ethical
requirements for anonymity. None-the-less, we sincerely thank all who have made this project
possible.
We thank the European Union for co-funding the project and providing support throughout the
duration of our work, specifically the EU officers who have supported the project team.
Stakeholder contributions have undoubtedly shaped the project and provided a significant resource
for the research team throughout the project. Without this contribution, the project itself and the case
study report would be a very different product. As such, we are especially grateful to all of the
professional and lay stakeholders in several countries who have contributed to the Stakeholder
Advisory Panels’ (SAPs) meetings and interviews and so enabled stakeholder involvement in the
‘Demonstration HTA’. It has been our pleasure and privilege to work with so many stakeholders from
diverse lay and professional backgrounds in the field of palliative care. All stakeholders are thanked
for generously giving their valuable time and sharing their experience and views about palliative care.
The Patient and Public Involvement Representatives who have undertaken a wider advisory role by
diligently attending meetings and making contributions on a monthly basis throughout the project as
well as assisting in dissemination activities are thanked for their continued and sustained input
throughout the project.
We would also like to thank service managers and others, including health care professionals and
members of the ethics committees, who have provided access to professional and lay stakeholders.
Without their understanding of the need for this case study, the work would not have been possible.
This case study report would also not be possible without the commitment of the project team who
has been dedicated to developing methods for Health Technology Assessment and applying these to
the case study. This team has included administrators who have provided vital support to sustain the
work of the whole team.
18
CHAPTER 1
Background and Introduction to the project
This chapter briefly summarises the background and rationale for the INTEGRATE-HTA project and
the ‘demonstration HTA’ case study. An overview of the INTEGRATE-HTA project aims and design is
given as well as an introduction to complex interventions and their characteristics. The rationale for
selecting palliative care for the ‘demonstration HTA’ case study is provided, the
intervention/technology described before the overall palliative care case study aims are stated and
the structure and content of this report is outlined.
Health Technology Assessment
HTA is an important tool to support health policy decision-making. IANHTA (2015) define HTA as “the
systematic evaluation of the properties and effects of a health technology, addressing the direct and
intended effects of this technology, as well as its indirect and unintended consequences, and aimed
mainly at informing decision making regarding health technologies.” IANHTA note that “HTA is
conducted by interdisciplinary groups that use explicit analytical frameworks drawing on a variety of
methods.” Similarly, the European network for Health Technology Assessment (EUnetHTA) define
HTA as a “…multidisciplinary process that summarises information about the medical, social,
economic and ethical aspects related to the use of a health technology in a systematic, transparent,
unbiased, robust manner. Its aim is to inform the formulation of safe, effective health policies that are
patient focused and seek to achieve best value” (EUnetHTA 2015). However, HTAs usually focus
primarily on cost and effectiveness, paying less attention to the assessment of wider issues, including
the ethical, socio-cultural and legal domains. There is also a need for more systematic methods for
including consideration of issues including context, implementation and patient preferences.
Additionally, the emphasis has been on summarising information which may not meet the needs and
preferences of decision makers for an integrated assessment of the available evidence. There is also
increasing recognition of the need for HTA to assess complex health technologies (the characteristics
of which are shown in Box 1), especially as changing disease patterns in European countries have
led to increasingly complex health technologies.
Complex health technologies
All health technologies (i.e. pharmaceuticals, disease management programmes) are, to varying
degrees, complex and operate within complex social systems. The starting point for considerations of
complexity in HTA is the MRC guidance on methods for developing and evaluating complex
interventions (Craig et al, 2008) which provides a useful summary of many of the relevant issues.
HTA in contrast to primary evaluation is concerned with summarising existing information about the
medical, social, economic and ethical aspects of a health technology to inform health policy/decision
making and may therefore require different considerations of complexity. For instance, in HTA
understanding how the intervention might interact with different health system contexts and settings
is of prime importance, whereas in primary evaluation controlling for underlying variation may be of
higher priority. Shiell (Shiell et al., 2008) highlights that complexity is a characteristic of the system
within which an intervention acts as well as being an inherent characteristic of an intervention itself.
Shiell describes complex systems as being adaptive to their local environment, as behaving non-
linearly and as being part of hierarchies of other complex systems. Shiell et al (2008) go on to
observe that much of what the MRC guidance describes under the label of ‘complex’ should more
correctly be defined as complicated, for instance interventions built up from a number of components
that may act independently or inter-dependently.
19
Thus whilst there are many common features between complicated interventions and complex
systems including; non-standardisation, multiplicity and interactions, HTA may have to recognise the
wider ramifications of complexity and attempt to make use of the insights gained from the complexity
science domain. Important aspects of complexity are displayed in Box 1.
Box 1.1 Aspects of complexity
Despite developments in recent years, contemporary HTA is currently sub-optimally equipped to
provide all the information that decision makers need to make critical decisions about what health
care to provide and how best to organize its delivery, especially in relation to the assessment of
complex technologies. In order to achieve a more structured, transparent, comprehensive, patient-
centred assessment of complex technologies (evidence-informed context based decision making),
policy-makers need HTA-results that bridge current methodological gaps to support their decision
making.
Aims of the INTEGRATE-HTA Project
The INTEGRATE-HTA project aims to adapt and develop concepts and methods for HTA to enable an integrated assessment of aspects of complex technologies as follows:
Assessment of effectiveness and economic, social, cultural, and ethical issues of complex technologies
Assessment of patient preferences and patient-specific moderators of treatment
Assessment of context, setting, and implementation
Integrating all issues in a comprehensive patient-centred assessment
Closing existing methodological gaps in the assessment of complex technologies
Design of the INTEGRATE-HTA Project
The design of the INTEGRATE-HTA project comprises the development of methods and concepts for
the assessment of complex technologies. These methods are applied in a case study in order to
evaluate their use for assessing complex interventions, and to demonstrate their application.
Case Study on Palliative Care – A ‘Demonstration HTA’
This ‘demonstration-HTA’ case study has been developed to illustrate the application of some of the
concepts and methods developed within the INTEGRATE-HTA project to palliative care as one
example of a service that is highly complex.
Number of groups or agents acting with intention in the system
Number and nature of interactions between agents in the system
Nature of control within the system
Degree of variability in intention and response of agents in the system
Potential for adaptive behavior within the system
Degree of flexibility and co-evaluation of intervention and setting
Degree of historicity, time and path dependence
20
Rationale for the selection of palliative care for the case study
In keeping with global trends, internationally, European populations are ageing (Gomes et al., 2011).
People are living with more complex, chronic conditions (e.g. cardiovascular, respiratory disease and
diabetes) and a greater number of co-morbidities (Payne et al., 2008). Advances in medicine and
health technologies mean that greater numbers of people will survive with complex health and social
care needs. Palliative care is defined by The World Health Organization (WHO) (2015) as “an
approach that improves the quality of life of patients and their families facing the problems associated
with life-threatening illness, through the prevention and relief of suffering by means of early
identification and impeccable assessment and treatment of pain and other problems, physical,
psychosocial and spiritual.” It is estimated that 60 per cent of the 56 million people dying world-wide
each year could benefit from palliative care (WHO, 2012). (See appendix 4 for more details about the
global policy context for palliative care).
Palliative care is interdisciplinary and holistic in nature (i.e. addresses physical. psychological, social,
spiritual and emotional needs) and treats the patient and family as the ‘unit of care’ (Radbruch and
Payne, 2009). Hence palliative care consists of several interventions at different levels that interact
which each other. Palliative care aims to relieve suffering and improve quality of life for both patients
and families throughout a life limiting illness, not just at the end of life (Pastrana et al., 2008).
Palliative care is a dynamic and evolving concept, the meaning of which has changed over time (Gott
et al., 2013). Originally developed in England, historically, palliative care was provided towards the
end of life, primarily for patients with cancer. More recently, palliative care is provided for a much
wider patient population (for example, those with motor neurone disease (MND), heart failure,
HIV/AIDS, dementia) (Murray and Sheikha, 2008, Van Mechelen et al., 2013) and earlier in the
patient’s illness; sometimes concurrently with curative care (Temel et al., 2010).
These features indicate that palliative care is highly complex and consists of all the conceptual and
methodological challenges associated with complex technologies. Furthermore, palliative care also
has overarching relevance for future health care across Europe (see Appendix 5). As such, palliative
care is a relevant case study for the application of methodologies developed within the framework of
the INTEGRATE-HTA project.
Specialist palliative care as the focus for assessment
Palliative care is delivered by two distinct categories of health and social care professionals:
specialist and generalist palliative care providers. ‘Generalist palliative care’ providers, are those
health professionals who have not received accredited levels of training in palliative care provision
and, thus, are not deemed ‘specialists’, but who routinely provide health care for patients at the end
of their lives (Gott and Ingleton, 2011). Generalist palliative care providers may include General
Practitioners/family doctors, community nurses, nursing home staff, hospital staff and others who
have not undertaken specialist training in palliative care. ‘Specialist palliative care’ is provided by
teams of multidisciplinary palliative care professionals who have undertaken specialist training in
palliative care and work only within the field of palliative care (Payne, 2010).
The remit of the specialist role is to care for patients who require continuous or high levels of support,
and to focus upon the management of ‘complex’ cases and providing support to generalist
colleagues through partnership working. Specialist palliative care services offer a range of provision,
from a single specialist nurse to a comprehensive multidisciplinary team. Specialist palliative care
services have developed an array of different types of provision including: in-patient units – hospices,
hospital teams, community teams, out-patient clinics, day care, respite services, bereavement
support services, complementary therapies, counselling and psychological support, spiritual and
religious support.
21
The terms ‘specialist’ and ‘generalist’ are used to denote these different aspects of palliative care
provision, with the understanding that categorization of this nature is not as straightforward as the
terminology would imply, and that terms may be subject to differing interpretations across Europe.
The complexity of service provision offered by specialist palliative care teams combined with the level
of interaction and collaboration with generalist colleagues, identify specialist palliative care as a
suitable and appropriate complex intervention for this case study.
Models of Palliative Care
Current HTA in the field of palliative care has focused on the evaluation of specific individual
technologies for specific conditions (for example specific pain management options for specific
patient groups which a specific type of pain) or broad reviews of models of care (for example recent
New Zealand review of evaluations of “models of care” Arora et al., 2011). The project proposal
identified models of care as a suitable focus because decision makers need to determine the most
appropriate ways to deliver palliative care services.
The term ‘model’ of palliative care is defined with regard to its structure only as ‘who delivers (e.g.
professionals, paid carers) the intervention (specialist or generalist palliative care), where (setting –
e.g. hospital), to whom (care recipients), when (i.e. timing and duration), how (e.g. face to face) and
for what purpose (i.e. expected outcomes)?” The focus is on structural models of palliative care.
Stakeholder involvement to determine the focus of the HTA
Stakeholders (i.e. people who are likely to experience the consequences of the HTA (Guba and
Lincoln, 1985)) have been integral part of the case study (see Appendix 6) and assisted in the
identification of key issues in palliative care (see Chapter 2). Stakeholder involvement in the project
and a review of review level evidence about models of palliative care (see Chapter 2) led to a focus
on two major models of palliative care: home based palliative care and reinforced home based
palliative care which are described in the following text.
Non-reinforced models of home based palliative care (HBPC)
Gomes et al’s (2013 p10) definition of home based palliative care (HBPC) is used to describe non-
reinforced models in this case study. HBPC has four elements and can be summarised as
interventions provided directly to patients:
1. “primarily with a severe or advanced disease (malignant or non-malignant), no longer responding to curative/maintenance treatment or symptomatic (or both) or their family caregivers, or both
2. aiming to support patients or their family caregivers, or both outside of hospitals and other institutional settings as far as possible to enable patients to stay at home and in which it was clear the majority of service contacts were stablished while the patients were at home
3. providing comprehensive specialist or intermediate palliative/hospice care as defined in a systematic review by Higginson in 2003 (see Appendix 7)
4. providing comprehensive care aimed at different physical and psychosocial components of palliative care”. (Gomes et al., 2013, p10)
22
Non-reinforced models of HBPC primarily focus on patients, though aspects of care may also be
directed towards caregivers on an ad hoc basis rather than as specific planned intervention. This is
because palliative care is underpinned by a philosophy that views both the patient and caregiver as a
‘unit’ of care. However, the attention given to informal caregivers is often dependent on the delivery
agent’s recognition of the informal caregiver’s needs. Therefore, informal caregiver’s experience is
inconsistent.
Reinforced models of home based palliative care (rHBPC)
Gomes et al’s (2013) definition of ‘reinforced models of home based palliative care’ (rHBPC) is used
to mean “existing home palliative care with an additional component of caregiver support” (p19).
According to our definition, reinforced models of home care will always include an intentional and
explicit attempt to support informal (i.e. not professional) caregivers in addition to the care given to
patients. ‘Support’ is broadly defined by Gomes et al as any psycho-educational intervention aimed at
providing assistance to caregivers (e.g. individual or group counselling, education, advice or respite
services). Hence, this support may be more active in nature, aiming to prepare the caregiver for the
patient care to come, by teaching the necessary skills, or it may be reactive in nature, aiming to
address psychological health burden of the lay caregiver resulting from care. Some reinforced
models are underpinned by specific theories, such as stress-coping theories (McMillan and Small,
2007), while others may not be based on theory (Harding et al., 2002).
It is worth noting that the term ‘reinforced’ care arose from the typology of services identified by
Gomes et al (2013) and is not one that is widely used or recognised or used in practice. It is also
worth noting that many overlaps exist between reinforced and non-reinforced home-based models of
palliative care which only differ in that reinforced models provide explicit support to the caregiver.
England as the context for the palliative care case study
Internationally diverse contexts for and understandings of, palliative care exist as well as variation in
service development (see Appendix 8). Palliative care is in various stages of development across
Europe and provision differs substantially (Centeno et al., 2013). Some European countries have well
developed palliative care services (e.g. the Italy, Poland and Germany), whilst others have much less
provision (Centeno et al., 2013). As palliative care began in England and services there are
comparatively well-developed palliative care services (Centeno et al., 2013). A difference in the
provision of palliative care services is perhaps not surprising as European countries have different
legal, ethical, cultural and social contexts.
Given the variability in services across Europe, this HTA focuses on England and has been informed
by issues from stakeholders in each of the countries involved in the project (England, Germany, Italy,
Lithuania, the Netherlands, Norway and Poland). We report individual country results from
stakeholder consultations in the appendices of this case study report. England was selected as the
context for this ‘demonstration HTA’ case study because services are well developed in England and
the issues raised by stakeholders in England reflected those raised by stakeholders in other
countries involved in the project. The intention is to ensure that the project findings have international
relevance by focusing on issues that are of shared concern to stakeholders across the European
countries involved in the project.
23
About this case study report
Purpose of the Case Study Report
This case study report will:
1. Show the feasibility and value of applying the concepts and methods developed within the INTEGRATE-HTA project using models of home based palliative care as a specific example of a complex intervention.
2. Summarise the evidence comparing reinforced and non-reinforced models of home based palliative care through the application of new concepts and methods for HTA that enable:
Assessment of effectiveness and economic, social, cultural, legal and ethical issues of complex technologies
Assessment of patient preferences and patient-specific moderators of treatment
Assessment of context, setting, and implementation
Integrating all issues in a comprehensive patient-centred assessment
The overall case study aims to provide a synthesis of a broad range of evidence that can be used by those commissioning and developing palliative care services to support holistic, patient-centred evidence-based decision-making in a complex field which requires a shared understanding of the issues involved.
Audiences for the ‘Demonstration HTA’
The final results of the INTEGRATE-HTA project include newly developed frameworks or guidance
for choosing an appropriate method from several existing options. The primary purpose of this case
study was to demonstrate the application of the products being developed within the INTEGRATE-
HTA project to palliative care as a specific example of a complex intervention (in this case, a
comparison of reinforced and non-reinforced home care models). As well as providing valuable
feedback for the subsequent development and refinement of HTA methods within the INTEGRATE-
HTA project, this report provides relevant information for two different audiences; both the HTA
community and the palliative care community. At the end of the report we summarise the useful
messages and learning both audiences.
Structure of Case Study Report
The case study report is structured using the INTEGRATE-HTA model. The INTEGRATE-HTA model
was developed to enable integration of relevant assessment aspects (Figure 1.1). The process model
is based on the results of a systematic review on integration. The review found that no single
integration approach covers all aspects of a complex HTA. Critical assessments, synthesis of
evidence, creating logic models and decision support tools, such as multi-criteria decision analysis
(MCDA), all have integrative features. The INTEGRATE-HTA model, used to structure this case
study report demonstrates how it may be possible to combine elements of these different approaches
in the context of HTA. The INTEGRATE-HTA model consists of the following six steps:
1. Definition of the HTA objective and technology
2. Creation of a logic model to define evidence needs
3. Evidence collection and preparation
4. Evidence processing and integration
5. Managing the complex evidence
6. Decision-making
24
Figure 1.1 The INTEGRATE-HTA model for the integrated assessment of complex technologies
(Wahlster et al., 2016)
25
A summary of each step of the process model is presented within the following chapters of this report
(Chapter 2-7). Chapter 2 presents an overview of steps 1 and 2 as these provide background information
and stages involved in the preparation for evidence collection. Steps 3, 4, and 5 are presented in
chapters 3, 4 and 5 respectively. Chapter 6 presents the lessons learned from the case study, the
conclusions and recommendations for both HTA researchers and palliative care.
References
ARORA, N., STANDFIELD, L., & WESTON A (2011) A. Systematic review of systems of palliative Care HSAC Report; 4 (22).
CENTENO, C et al., (2013) "EAPC Atlas of Palliative Care in Europe 2013 - Full Edition" Milano: EAPC (European Association for Palliative Care), http://hdl.handle.net/10171/29291.
Craig, P., Dieppe, P., Macintyre, S., et al (2008) Developing and evaluating complex interventions: the new Medical Research Council guidance. British Medical Journal, 337(sep29 1), a1655-a1655. doi: 10.1136/bmj.a1655
GUBA, E and LINCOLN, E. Fourth Generation Evaluation. SAGE, Newbury Park (CA), 1985.
EUnetHTA What is Health Technology Assessment (HTA)? Available at http://www.eunethta.eu/faq/Category%201-0#t287n73 (2015 accessed 10 January 2015).
GOMES, B., COHEN, J., DELIENS, L., HIGGINSON, IJ. International trends in circumstances of death and dying. In Gott M, Ingleton C, editors. Living with ageing and dying: palliative and end of life care for older people. Oxford: Oxford University Press; 2011. pp. 3–18.
GOTT, M., INGLETON, C., GARDINER, C., et al (2013) Exploring the transition from curative care to palliative care: a systematic review of the literature (phase 1).
GOTT, M., & INGLETON, C. (Eds.). (2011). Living with ageing and dying: palliative and end of life care for older people. Oxford University Press.
HARDING, R., LEAM, C., PEARCE, A., et al (2002) A multi-professional short-term group intervention for
informal caregivers of patients using a home palliative care service. Journal of Palliative Care, 18(4): 275-81.
INAHTA (2015) Health Technology Assessment (HTA) in HTA Glossary.net [online] Available from:
http://htaglossary.net/HomePage Last [Accessed 27 September 2015]
MCMILLAN, SC & SMALL, BJ (2007) Using the COPE intervention for family caregivers to improve symptoms
of hospice homecare patients: a clinical trial. In Oncology Nursing Forum, 34(2): 313-321.
MURRAY, S & SHEIKHA, A (2008) Palliative care beyond cancer: cancer for all at the end of life. British
Medical Journal, 336: 958-9.
PASTRANA, T., JÜNGER, S., OSTGATHE, C., et al (2008) A matter of definition–key elements identified in a
discourse analysis of definitions of palliative care. Palliative Medicine, 22(3): 222-232.
PAYNE, S (2010) EAPC Task Force on Family Carers White Paper on improving support for family carers in palliative care: part 1. European Journal of Palliative Care. 17(5):238-45.
RADBRUCH, L & PAYNE, S (2009) EAPC Board of Directors White Paper on Standards and norms for hospice and palliative care in Europe: part 2. European Journal of Palliative Care, 17(1): 22-33
SHIELL, A., HAWE, P & GOLD, L (2008) Complex interventions or complex systems? Implications for health economic evaluation. British Medical Journal, 336(7656): 1281.
TEMEL, JS., GREER, JA., MUZIKANSKY, A., et al (2010) Early palliative care for patients with metastatic non–small-cell lung cancer. New England Journal of Medicine, 363(8): 733-742.
VAN MECHELEN, W., AERTGEERTS, B., DE CEULAER, K., et al (2013) Defining the palliative care patient: a systematic review. Palliative Medicine, 27(3): 197-208.
WAHLSTER, P., BRERETON, L., BURNS, J., HOFMANN, B., MOZYGEMBA, K., OORTWIJN, W., PFADENHAUER, L., POLUS, S., REHFUESS, E., SCHILLING, I., VAN HOORN, R., VAN DER WILT, G.J., BALTUSSEN, R., GERHARDUS, A. (2016) Guidance on the integrated assessment of complex health technologies - The INTEGRATE-HTA Model [Online]. Available from: http://www.integrate-hta.eu/downloads/
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CHAPTER 2
Steps 1 & 2 of the HTA model: The HTA Objectives & Technology & model creation to
define evidence needs
This chapter addresses key elements included in Steps 1 and 2 of the INTEGRATE-HTA model which
identify the scope of the case study (i.e. the plans for the case study including the focus of the work and
what will be included and excluded) (Figure 2.1). Step 1 defines the HTA objective and presents
preliminary definitions of the technologies of interest. Step 2 results in the creation of a logic model,
which defines evidence needs for the HTA. Some elements of Step 1 have already been presented in
Chapter 1 (i.e. the HTA researchers and selection of palliative care for the assessment). The remaining
elements of Steps 1 and 2 relate to 3 key processes; 1) the context for the HTA 2) definition & refinement
of the HTA objective (as the result of SAPs & evidence review) 3) development of the logic model. As the
processes used to develop the project scope were iterative, for clarity, the elements addressed in Steps 1
and 2 are considered together in this chapter. Collectively, Steps 1 and 2 of the INTEGRATE-HTA model
enabled:
1) consideration of the context for the HTA, particularly with regard to decision makers and the functional requirements of the decision making bodies in England
2) definition and refinement of issues and outcomes relevant to the HTA objective which resulted in the identification of reinforced models of palliative care (rHBPC) as the focus for the case study. This occurred primarily as a result of drawing together information from
a) stakeholder consultation in seven countries (England, German, Italy, Lithuania, the Netherlands, Norway and Poland) to identify key issues in palliative care (i.e. the Stakeholder Advisory Panels (SAPs) identified in Steps 1 & 2)
and
b) a review of review level evidence to identify and define specific technologies of interest
3) development of a logic model template (Step 1) and completion of the logic model which guided evidence collection for some aspects of the case study (Step 2).
27
Figure 2.1 Step 1 & 2 of the INTEGRATE-HTA model
(Wahlster et al., 2016)
28
The context for the HTA
As previously indicated, England was selected as the context for the case study and the HTA centres on
England because palliative care policy and services are comparatively well-developed in relation to other
countries in Europe (Centeno et al., 2013). (For more information on the policy context for palliative care
and informal caregivers in England see Appendix 9). As such, contextual information will relate primarily
to England where health care policy includes an End of Life Care Strategy (DH, 2008) and there is
recognition that patients requiring palliative care need to be supported in complex care systems (King’s
Fund, 2011). Although the INTEGRATE-HTA project was not commissioned by Health Technology
Assessment decision makers, for the purpose of the demonstration-HTA an overview of the HTA process
in England is provided in Appendix 10. Appendix 11 identifies key decision makers in England; the
functional requirements of the National Institute for Health and Care Excellence (NICE) (the main national
HTA commissioning agency) in England and information about commissioning palliative care services in
England.
Logic Model Template
A logic model is “… a graphic description of a system…designed to identify important elements and
relationships within that system” (Craig et al., 2008). The graphic representation of concepts, theories,
causal pathways and context is helpful in understanding the various components of complex
interventions and their relationship to the various levels of outcomes. Traditionally, logic models have
been used for programme evaluation purposes but in recent years, they have become a popular tool
when doing research synthesis of complex interventions. By making underlying theories and assumptions
about causal pathways explicit, a logic model provides a framework that guides conceptualisation of the
intervention and question formulation, helps to identify sources of heterogeneity and plan data synthesis.
Logic models are thus useful at every stage of the review/HTA process and can be of great value when
interpreting results and identifying areas for further research. Importantly they increase transparency of
assumptions amongst the research team and can make results more accessible to various stakeholders.
Although there are a few published examples of logic models in systematic reviews and HTAs, at the
outset of the INTEGRATE-HTA project there was no specific guidance on how to develop an appropriate
logic model. Figure 2.2 shows the system-based logic model template developed in Step 1 (see Rohwer
et al., 2015). This template allows clear identification of the participants, intervention, comparator,
outcomes, context and implementation, thus providing a comprehensive description of components of the
health care system. The logic model template is informed by evidence from a variety of sources e.g.
literature, stakeholders, discussions within the research team. Its aim is to clarify and make transparent
the assumptions of the research team regarding the complex intervention and its effects.
29
Figure 2.2 The Logic Model Template
30
Definition and refinement of the HTA objective
The initial project proposal identified models of palliative care service provision as a possible area for
interest for the palliative care case study. The initial intention (in Step 1) was to compare specialist and
generalist models of palliative care. Stakeholder consultation took place to determine issues of
importance to palliative care stakeholders concurrently with a review of review level evidence about
models of palliative care to confirm the feasibility of comparing specialist and generalist models of
palliative care in the case study. The HTA objective was refined after stakeholder consultation and a
review of review level evidence. Refinement of the HTA objective resulted in a focus on specialist
models of palliative care, specifically reinforced (rHBPC) and non-reinforced models of home based
palliative care (HBPC).
Stakeholder involvement at the outset of the project
Stakeholder involvement in both HTA and palliative care research is supported internationally (HTAi,
2014, Bradburn & Maher, 2005), to ensure that important issues are identified and addressed by
researchers. The INTEGRATE-HTA team’s commitment to stakeholder involvement ensured this has
been an integral part of the methods used in Steps 1 and 2. Methods of stakeholder involvement vary,
with some countries favouring qualitative research approaches (e.g. the Netherlands) and others (e.g.
England) favouring a consultative approach.
Stakeholder Advisory Panels (SAPs)
Although England is the context for the case study, to increase the international relevance and potential
value of the case study we involved stakeholders in seven countries (England, Germany, Italy, the
Netherlands, Norway, Lithuania and Poland). At the outset of this project, stakeholders were used as
advisors to assist in the identification of key issues relating to palliative care in each country.
Stakeholders contributed to ‘stakeholder advisory panels’ (SAPs) in their own country. The term ‘panel’
refers to the collective information provided by individuals or groups (not the establishment of a ‘panel’ or
group of stakeholders) as patients with palliative care needs and professionals cannot always attend
face-face meetings, especially when stakeholders are geographically dispersed. The SAPs provide the
opportunity for stakeholders (e.g., clinical experts, academics, patients and the public) to contribute to
the INTEGRATE-HTA project at a national level to enhance the international relevance of the project
findings. Patients, family carers, clinicians and academics have different types of expertise in palliative
care. As a result of their experience in palliative care, consultation with these ‘expert’ lay and
professional stakeholders helps to build agreement among various stakeholders as to what the relevant
questions seem to be. As such, stakeholder consultation can help researchers to ask the right questions,
therefore assisting in ensuring that the results of the study are useful to both service users and
providers. Other stakeholders with an interest in, but perhaps limited experience of, palliative care are
also valuable advisors because palliative care is a public health issue of relevance to the whole of
society.
Appendix 12 provides an overview of the stakeholder involvement across all seven countries. A total of
132 stakeholders (82 professionals and 50 ‘lay’ people) participated in individual face- to- face or
telephone meetings/interviews, group meetings or focus groups. Two main approaches were used to
identify stakeholders’ views of key issues in palliative care which informed project development (Steps 1
and 2 of the INTEGRATE-HTA model), thus ensuring that the critical concerns of all stakeholders are
considered can assist decision-making. Using a consultative approach based on either the National
Institute for Health and Care Excellence (NICE) (2012) guidance or a qualitative approach, stakeholders
in each of the seven countries (England, Germany, Italy, The Netherlands, Norway, Lithuania and
Poland) provided information and advice that assisted in identifying the focus of the project. Adult (i.e.
aged 18 years and over) ‘lay’ (i.e. patients, family, friends and carers) and ‘professional’ stakeholders
31
(including palliative care service commissioners, clinicians, practitioners and academics) played a key
advisory role from the outset of the project.
Consultations with stakeholders in seven countries highlighted a range of similar issues. Thematic
analysis was used to identify key issues across countries. Four themes were identified as common
across all seven countries: recognition of palliative care needs; funding; the organisation of palliative
care and professionals’ training. A total of twenty-three issues that were common to three or more
countries were identified. These issues included the need to increase home care provision in four
countries (England, Italy, Norway and Poland) and provide training and support to family carers in four
countries (England, Italy, Lithuania and Poland). (See Appendices 13-19 for further details about the
SAPs across all seven countries).
A review of review level evidence to identify and define specific technologies of interest
At the outset of the project, the complexity of palliative care service provision indicated that a comparison
of specialist and generalist palliative care provision would provide a suitable and appropriate complex
intervention for this case study. Although broad reviews of models of care exist, for example a review
evaluating “models of palliative care” in New Zealand (Arora et al., 2011), more work is needed in terms
of evaluating effectiveness and wide issues for both specialist and generalist palliative care service
provision and uptake. Harding & Higginson (2005) highlighted the need to examine what constitutes
feasible, accessible, and effective palliative care, and how to develop such services as a problem that
remains to be resolved.
Having identified the possible intervention and comparator of interest in the project proposal, a
systematic review of review level evidence was undertaken to identify the range of models of palliative
care that exist; their advantages and disadvantages and which components are likely to be most
effective for which patient groups under what circumstances. A review of systematic and narrative
reviews according to the preferred reporting items for systematic reviews and meta-analyses (PRISMA)
guidelines was completed. Given the breadth of the INTEGRATE-HTA project aims, the team decided
not to limit the assessment to specific outcomes, especially as Arora et al’s (2011) review examines only
eight outcomes. The limitation of this is apparent given that Stiel et al’s (2012) systematic review of
outcome assessment instruments in palliative and hospice care reported over 500 instruments existed to
measure outcomes that could be assigned to four domains relating to patient, family members or
caregivers, staff members / professionals and the health care system.
Comprehensive searches were conducted of 7 databases were completed to identify systematic and
narrative review level evidence about models of palliative care for any palliative care adult patient group
(defined as people aged 18 and over) with life-limiting illnesses. Data sources including MEDLINE,
EMBASE, PsycINFO, CINAHL, Cochrane Library (Cochrane Databases of Systematic Reviews and
Database of Abstracts of Reviews of Effects), Web of Science and ASSIA were searched for reviews
published between 2000 and 2014. Reference lists of included reviews and searches of Google were
completed to identify further reviews. Hand searches were not undertaken. A total of 17241 papers
identified. Two reviewers independently screened titles and abstracts for relevance using pre-
determined inclusion criteria. Two reviewers independently extracted data for each included study.
Methodological quality was assessed using the AMSTAR tool1. Narrative synthesis was undertaken. A
range of models of palliative care have been described and evaluated in 18 medium - high quality
reviews of specialist palliative care. Most were narrative reviews based on non-randomised studies.
The results showed that little has been written about generalist models of palliative care. Most evidence
appears to relate to specialist models of palliative care, notably home based palliative care, although
some models are delivered across care settings (e.g. hospice, hospital, home). Although individual
reviews highlighted a number of potential advantages, and a few disadvantages, of models of palliative
32
care, heterogeneity in descriptions of models of palliative care and poor quality of reporting about the
components of both intervention and comparator models is a major barrier to the evaluation of models of
palliative care.
The heterogeneous nature and the poor quality of reporting about the components of models makes it
difficult to draw any conclusions about which models / components of models are most appropriate for
which patient groups under what circumstances. Indeed, the under-reporting about the components of
both intervention and comparator model is a major barrier to the implementation of models of palliative
care. Limited evidence exists about the cost-effectiveness of each model. (For further information about
the review of review level evidence, see Appendix 20).
Identification of home based palliative care for application of INTEGRATE-HTA methods:
a) Identifying home based palliative care for the case study
The evidence base within the review of reviews alone was insufficient to inform future reviews /
testing of the methods developed in the INTEGRATE-HTA project. Despite the limitations of the
evidence base for informing decision making about future potential service models, most of the
available evidence related to home care. Whilst completing the review of reviews, we also consulted
with lay and professional palliative care stakeholders in seven countries who acted as advisors to
inform the development of the project scope. These stakeholders provided information about the key
issues in palliative care. These key issues helped to inform the decision to use reinforced and non-
reinforced models of home based palliative care (as described in Gomes et al’s (2013) Cochrane
review) as the focus for the application of the new concepts and models developed in the project.
(See pages 22 and 23 for definitions of reinforced and non-reinforced models of home based
palliative care). The rationale for the choice of home care models related to: i) evidence about patient
preferences of home care/ death; ii) policy initiatives to increase availability of home palliative
care/home death across several countries involved in the project; iii) the volume & quality of evidence
identified in the reviews; iv) reflecting the philosophy of palliative care re patient and caregiver as a
unit of care; v) explicitly addressing one of the key issues raised by stakeholders in several countries.
b) Relevant Issues
The focus on reinforced and non-reinforced models of home based palliative care enabled the project
to explicitly address one of the key issues raised by stakeholders in several countries (i.e. the need
for caregiver training/support) as well as many of the other wider issues raised by the SAPs.
c) Relevant Patient Groups
The focus on adults aged 18 years or older and their families was considered appropriate when
assessing reinforced and non-reinforced models of home based palliative care. Whilst the choice of
reinforced models of home palliative care as the intervention of interest meant that greater attention
would be afforded to carer issues, the team recognised that caregiving for children presents very
different problems than that of an adult population (Groh et al., 2014).
d) Outcomes
No attempt was made to limit the outcomes of interest in the SAPs or in the review of reviews as the
intention was to identify key issue of importance to stakeholders and suitable interventions for use in
the case study respectively. The decision was upheld on reassessment of the outcomes of interest for
reinforced and non-reinforced models of home based palliative care as it was important to ensure that
all relevant aspects of these interventions, and therefore all potential outcomes, could be used in the
HTA (i.e. including outcomes related to effectiveness, legal issues etc).
e) The case study research question
33
As a result of the review of reviews and advice from stakeholders in the seven European countries,
the question for the case study was revised to specify reinforced and non-reinforced models of home
based palliative care as the intervention and comparator respectively:
Are reinforced models of home based palliative care acceptable, feasible, appropriate, meaningful,
effective, cost-effective model for providing patient-centred palliative care [compared to non-reinforced
(i.e. ‘usual’) models of home based palliative care] in adults (defined as those aged 18 years old and
over) and their families? (See Appendix 1 for definitions of the key terms used in the research question).
Creating a specific logic model for reinforced (rHBPC) and non-reinforced home palliative care (HBPC)
Having developed the logic model template, using iterative processes in Step 1, we initially completed
this to produce a system-based logic model for specialist and generalist palliative care in England. On
completion of the stakeholder consultation and review of review level evidence about models of palliative
care, in step 2, a specific logic model was developed for reinforced (rHBPC) and non-reinforced models
of home based palliative care (HBPC) as the intervention and comparator of choice for the case study in
the demonstration HTA (see Figure 2.3).
The logic model template for reinforced (rHBPC) and non-reinforced models of home based palliative
care (HBPC) was completed using an iterative process in which the developers of the logic model and a
researcher with experience of working in palliative care drew on the palliative care policy (England
specific), literature identified through ongoing, non-systematic, searches, reflective thoughts and
information from the initial stakeholder involvement.
Emphasis is required on informal caregivers as participants in rHBPC; interventions include an additional
component of caregiver support (additional to the ‘usual’ home based palliative care intervention). The
additional component of caregiver support found in reinforced models of home based palliative care
affords explicit attention on the informal caregiver as a care recipient. There is also greater focus on
caregiver outcomes for reinforced models of home based palliative care. The logic model also includes
specific reference to informal caregivers within the sections on context and implementation. Further
explanatory text for this model can be found in Appendix 21.
This logic model provided the ‘starting’ place for further investigation of the evidence base relating to
reinforced and non-reinforced models of home based palliative care.
34
Figure 2.3 Logic model of reinforced and non-reinforced home-based palliative care
Implementation
Policy o Quality of care and service organisation
strategies
o Financing/Reimbursement strategies
Financing o Public (e.g. taxation; insurance) o Private/ self-funding o Third sector/charity
Organisation and structure o Public sector
Context
Geographical
o European Union
o Urban vs. rural
Epidemiological
o Cancer focused palliative care
o Other diseases
Socio-cultural
o Ethnicity
o Religion
o Individual patient preferences
o Family and community preferences
Socio-economic
o Education
o Wealth
o Housing
Ethical
o Autonomy
o Sanctity of Life
o Beneficence
o Non-maleficence
o Justice
Legal
o Mental capacity act
Participants: Patients: adults with life limiting conditions (malignant and non-malignant) receiving palliative care at home
Lay caregivers: family members of patients or others (friends, neighbours) who may take on the role of lay
caregiving (≥18 years)
Intervention and comparison: Reinforced and non-reinforced home-based palliative care Intervention theory
holistic approach to improve quality of life and to enable a good death for patient
aim to allow the patient to be treated for and die at home, if desired
(Reinforced) explicit, structured support for the lay caregiver to alleviate burden due to caregiving
Intervention design Components:
Services addressing physical, psychological, social
and spiritual needs of patients
(Reinforced) Services explicitly providing
psychosocial or psychoeducational support to lay
caregiver
o Active and reactive support
Execution:
Timing, duration and frequency
May commence at any time from diagnosis to end of life and bereavement
Models of transition to palliative care e.g. concurrent palliative and curative care; palliative care upon cessation of curative care
Intervention delivery Delivery mechanisms:
Face-to-face /distant (telephone, online)/mixed
Individual/group/patient-carer dyad/mixed
Outcomes1,2
Intermediate outcomes Process outcomes:
Quality of care
Hospitalisation
Reach
Professional caregiver outcomes Surrogate outcomes (of patients and carers)
Coping
Mastery
Self-efficacy
Health outcomes Patients:
Quality of life
Physical well-being (reduced symptoms)
Psychological well-being
Spiritual well-being
Good death/achieving preferred place of death
Survival
1includes short-, medium-, and long-term outcomes 2includes proxy outcomes (need to be indicated)
(Key: Reflective thoughts Policy
literature Experts)
(Key: Reflective thoughts Policy literature Experts)
35
Summary of Step 1 and Step 2
Step 1 and 2 resulted in the identification of the following HTA question:
Are reinforced models of home based palliative care acceptable, feasible, appropriate, meaningful,
effective, cost-effective models for providing patient-centred palliative care [compared to non-
reinforced models of home based palliative care] in adults (defined as those aged 18 years old and
over) and their families?
References
ARORA, N., STANDFIELD, L & WESTON, A. (2011) A. Systematic review of systems of palliative Care HSAC Report; 4 (22).
BRADBURN, J AND MAHER, J. (2005) User and carer participation in research in palliative care. Palliative Medicine, 19(2): 91-92.
CENTENO, C et al., (2013) "EAPC Atlas of Palliative Care in Europe 2013 - Full Edition" Milano: EAPC (European Association for Palliative Care), http://hdl.handle.net/10171/29291.
CRAIG, P., DIEPPE, .P, MACINTYRE, S et al (2008) Developing and evaluating complex interventions: the new Medical Research Council guidance. British Medical Journal, 337(sep29 1), a1655-a1655. doi: 10.1136/bmj.a1655.
DEPARTMENT OF HEALTH (2008) End of Life Care Strategy: promoting high quality care for all adults at
the end of life. London: England.
GROH, G., FEDDERSEN, B., FÜHRER, M., et al (2014) Specialized home palliative care for adults and children: differences and similarities. Journal of Palliative Medicine, 17(7): 803-810.
HARDING, R & HIGGINSON, IJ. (2005) Palliative care in sub-Saharan Africa. Lancet, 365(9475):1971–5.
KINGS FUND (2011) Issues facing commissioners of end-of-life care. Kings Fund: London
NATIONAL INSTITUTE FOR HEALTH AND CARE EXCELLENCE (NICE) ‘Methods for the development
of NICE public health guidance' (3rd
edition) www.nice.org.uk (2012, accessed 12 December 2014).
ROHWER, A., PFADENHAUER, L., BURNS, J et al (2015) Use of logic models in systematic reviews and health technology assessments of complex interventions. Journal of Clinical Epidemiology (submitted for publication).
STIEL, S., PASTRANA, T., BALZER, C et al (2012) Outcome assessment instruments in palliative and hospice care—a review of the literature. Supportive Care in Cancer, 20(11): 2879-2893
WAHLSTER, P., BRERETON, L., BURNS, J., HOFMANN, B., MOZYGEMBA, K., OORTWIJN, W., PFADENHAUER, L., POLUS, S., REHFUESS, E., SCHILLING, I., VAN HOORN, R., VAN DER WILT, G.J., BALTUSSEN, R., GERHARDUS, A. (2016) Guidance on the integrated assessment of complex health technologies - The INTEGRATE-HTA Model [Online]. Available from: http://www.integrate-
hta.eu/downloads/
WHO 2015 Global Health Observatory data NCD mortality and morbidity [Available at http://www.who.int/gho/ncd/mortality_morbidity/en/
36
CHAPTER 3
Step 3 – Evidence Collection
Overview of Step 3
This chapter presents Step 3 of the INTEGRATE-HTA model (see Figure 3.1), specifically the
evidence reports resulting from the application of guidance developed within the INTEGRATE-HTA
project to assess reinforced and non-reinforced home palliative care. The specific logic model guiding
data collection for some aspects of evidence collection was presented in Step 2. Therefore, limited
attention will be given to the specific logic model in this chapter. An overview of the methods used to
collect evidence is provided in this chapter along with details of stakeholder involvement where this
occurred. A more detailed account of the methods used is provided in the appendices to the case
study and guidances developed in the INTEGRATE-HTA project. Evidence is presented from the
assessment of effectiveness, economics, ethics, socio-cultural, legal aspects; moderators of
treatment outcome, patient preferences, context and implementation. Within each evidence report,
reflections on issues of stakeholder involvement and the application of the methods developed are
included. Reflections on complexity and integration are drawn together at the end of the chapter.
37
Figure 3.1 Step 3 of the INTEGRATE-HTA Model
(Wahlster et al., 2016)
38
Evidence Reports
The effectiveness of home and reinforced home based palliative care
Background
Much evidence shows a preference among many receiving palliative care for care and death at home
(Bell 2009). Interventions should, therefore, reflect this preference by aiming to deliver care directly to
patients and informal caregivers outside the hospital or other institutional settings as far as possible,
and enable patients to remain at home if desired (Gomes, 2013a). Gomes et al (2013b) published the
review “Effectiveness and cost-effectiveness of home palliative care services for adults with
advanced illness and their caregivers” in the Cochrane Library in 2013. The objectives of the review
were to 1) quantify the effect of home palliative care services for adult patients with advanced
illnesses and their family caregivers on patients’ odds of dying at home; 2) examine the clinical
effectiveness of home palliative care services on other outcomes for patients and their caregivers
such as symptom control, quality of life, caregiver distress and satisfaction with care; 3) compare the
resource use and costs associated with these services; and 4) critically appraise and summarise the
current evidence on cost-effectiveness. Results of the review showed that home based palliative
care services were effective at increasing odds of dying at home, more than doubling the odds for
patients with illnesses such as cancer. With regard to other outcomes, results were less clear cut, as
heterogeneity in the home services offered, inconsistent estimates, a range of outcome measures,
variable study quality limited conclusions (Gomes, 2013b).
Care at home can be rewarding for those closest to the patient, including the informal caregiver
(Henriksson, 2013). It has also been shown in some studies, however, that such care at home may
worsen the burden of informal caregivers close to the patient, which could lead to increased physical,
psychological and social stress during care, as well as after the death of the patient (Sherman, 1998).
Gomes et al. classified interventions that offered an additional, explicit component of caregiver
support as reinforced home based palliative care. This evidence base was especially scant, as only
these four studies were identified, with limited evidence of effectiveness.
Aims
Given the importance of evaluating how existing services address this increased burden of informal
caregivers, we similarly sought to assess the effectiveness of reinforced home based palliative care
interventions, as compared to non-reinforced home based palliative care interventions, across a
range of patient- and informal caregiver-important outcomes.
Methods
We updated the Gomes et al (2013) systematic review with regards to their objectives 1) and 2) i.e.
1. quantify the effect of home based palliative care services for adult patients with advanced
illnesses and their informal caregivers on patients’ odds of dying at home;
2. examine the clinical effectiveness of home based palliative care services on other outcomes
for patients and their caregivers such as symptom control, quality of life, caregiver distress
and satisfaction with care.
We altered these objectives, as we were interested in comparing reinforced with non-reinforced
home based palliative care interventions. Reinforced models are the subset of home based palliative
care services that are the focus of this effectiveness assessment. Reinforced models of home based
palliative care include at least one component offering explicit support to the informal caregiver.
39
This support may take many forms (e.g. counselling to relieve burden or training to prepare informal
caregivers for their role).
Appendix 22 provides a detailed account of the methods used. We updated Gomes et al’s (2013)
original searches for systematic reviews of patient or cluster randomized controlled trials (RCTs);
patient or cluster controlled clinical trials (CCTs); controlled before and after studies (CBAs) or
interrupted time series studies (ITSs) (EPOC 2013). Six data bases were searched; Cochrane
Central Register of Controlled Trials (CENTRAL) MEDLINE, EMBASE, Cochrane Pain, Palliative and
Supportive Care Trial Register, CINAHL and Health Technology Assessment (HTA). Databases were
searched starting from either April 2010 or November 2012, depending on the Gomes et al. search
strategy, until November 2014 for primary studies focusing on reinforced and non-reinforced home
based palliative care for adults with life limiting conditions and their informal caregivers (both aged >
18 years). We categorised outcomes from included studies as follows:
Table 3.1 Outcomes found in included studies
Patient Outcomes
Caregiver outcomes
Pain
Symptom control
QoL
Psychological health
Death at home
Hospitalisation
Response (e.g. coping, preparedness, mastery, etc.)
Satisfaction with care
QoL
Psychological health
Response (e.g. coping, preparedness, mastery, etc.)
Satisfaction with care
Two reviewers independently assessed titles and abstracts of each study, excluding studies
according to study design, population, intervention or outcome. Subsequently, two reviewers
independently assessed the full text of potentially relevant studies. Detailed data were extracted
regarding the study design, population, intervention, comparison, relevant outcomes and results for
both studies originally included by Gomes et al (2013b) and new studies identified through the
updated search. Data extraction was performed using a data extraction form from the Cochrane
Public Health Group previously adapted for the use in reviews of complex interventions, and
subsequently adapted for this review of home-based palliative care services. Risk of bias was
assessed using criteria developed by the Cochrane EPOC Review group (Cochrane EPOC).
Evidence Synthesis
The INTEGRATE-HTA Guidance to assess effectiveness aspects (Burns et al., 2016) gives an
overview of existing methods for assessing the effectiveness of complex technologies and describes
under what circumstances these methods may be appropriate. It thus provides those performing
effectiveness assessments with a flexible framework for choosing how to synthesize methods. To
apply the framework, a reviewer should:
1. Conduct a comprehensive scope of the effectiveness assessment,
2. Gain a thorough understanding of the characteristics of available methods,
40
3. (Conditionally) Specify methods a priori.
Given that, for certain technologies, it may not be possible to know what methods are appropriate
before the relevant evidence is identified, the guidance also allows these initially defined methods to
be treated as conditional. In such a case, after the searches have been performed and the potentially
relevant studies have been identified, considering these further steps may be necessary:
4. Assess methodological and clinical heterogeneity in the identified evidence base,
5. Specify final decision on methods.
For more detailed information regarding this framework for selecting appropriate methods, see the
Burns, et al (2015) Guidance to assess effectiveness aspects. In: Lysdahl, KB, Mozygemba, K, Burns
J, Chilcott, JB, Brönneke, JB, Hofmann (Eds) (2016) 'Guidance for assessing effectiveness,
economic aspects, ethical aspects, socio-cultural aspects and legal aspects in complex technologies.
Based on steps 1 and 2 above, i.e. given the expected methodological and clinical heterogeneity of
the included studies, we decided a priori to forego meta-analysis. We created harvest plots, a method
that has been shown an effective, clear and transparent way to portray heterogeneous evidence,
especially in instances where meta-analysis may be less appropriate (Ogilvie, 2008; Turley, 2013;
Burns et al., 2016). We then used the harvest plots as a basis for a gap-analysis of the existing
literature, which was subsequently used to inform semi-structured expert consultations.
Harvest plots
Harvest plots allowed us to graphically and neatly display and summarise effectiveness for all
interventions and outcomes of interest. We arranged studies on a matrix in columns according to
direction of effect – increasing effect, no effect or decreasing effect, and in rows according to the
outcome category. Additionally, information regarding study design – represented by the height of the
bar, and where no statistical analysis was performed – indicated with a dotted border, was portrayed.
The colour of the bars designates whether that study was originally included in Gomes et al. (black)
or newly identified through our update of the Gomes et al. review (white). We created separate
harvest plots for patient and caregiver outcomes.
Subgroup Analyses
We planned a priori to additionally use harvest plots to explore effectiveness in subgroups. By
arranging studies according to factors that could potentially influence effectiveness, we planned to
assess whether, in the included studies, a trend in effectiveness possibly due to such a factor of
interest can be seen. We planned only to perform exploratory subgroup analyses, and therefore to
define specific analyses as relevant only after all included studies were identified.
Post-Review gap analysis and Expert Consultations
In many effectiveness reviews of complex interventions, including this one, reviewers forgo the use of
meta-analysis, deciding often instead for a narrative summary. In such cases, reviews may deliver
unclear results and conclusions, which do not provide policy makers with sufficient evidence for
decision-making. Such assessments may even lead to further questions regarding the effectiveness
of included models. In an attempt to address these issues, we used a gap analysis to identify such
knowledge gaps, which then informed post-hoc expert consultations, as described below.
Gap analysis – Defining a semi-structure for Consultations
As a review team, we sought to identify the knowledge gaps not addressed by or arising during the
review through an open discussion. All members of the team reviewed the harvest plots, and used
the time to discuss the results further.
41
Knowledge gaps arising from the discussions could, for example, pertain to overall effectiveness of
reinforced models of palliative care or trends in effectiveness based on post-hoc subgroup analyses.
After potential gaps were identified in this manner, the review team discussed how to prioritise and
incorporate them into semi-structured consultations.
Semi-structured consultations
Within the effectiveness assessment, four professional stakeholders from three countries, England,
Germany and the Netherlands, were consulted individually (See Appendix 23 for further details about
the stakeholders involved in the gap analysis). The aim was to explore the review results further and
discuss the relevant research gaps identified as outlined above. Each expert was asked to suggest
methodological or palliative care related issues that may have contributed to, or in the future could
help to, address these knowledge gaps. We obtained a waiver from the Ethics Committee of the
Ludwig-Maximilians-University in Munich, Germany, given that certain methodological criteria of the
expert consultations were met to ensure the anonymity of the participants.
Findings
Included studies
Gomes et al (2013b) originally included 23 studies. Upon reassessment, we excluded four of the
originally included studies, as they did not meet our review criteria. We excluded these for studies
because a relevant, definable intervention was not evaluated (n=2) (Gomez-Batiste, 2010; Ward,
1987), a partially non-palliative population was assessed (n=1) (Zimmer, 1985), and because of
scarcity in reporting methods and results (n=1) (Buckingham, 1978). Of the resulting 19 studies,
Gomes et al. (2013b) categorised four as reinforced, and we classified one additional study, originally
classified as a standard home based service, as reinforced. This discrepancy is explained by the
overlapping nature of home based and reinforced home based palliative care models, making a
distinction between the two difficult.
Our updated search, focusing on identifying home based and reinforced home based palliative care
interventions, identified 6072 records, which was reduced to 4876 unique records after de-
duplication. We excluded 4818 studies due to non-relevance at the title/abstract screening stage,
leaving 58 studies to be assessed at the full text screening stage. Of these, we assessed 48 as non-
relevant, leaving 10 newly identified studies, six home based and four reinforced home based
studies. The results of the five original and four newly included reinforced home based interventions
are included in this review. The relevant information is outlined in the flowchart in Figure A22.1 in
appendix 22. As there is substantial overlap between reinforced and non-reinforced home based
palliative care services, a problem exacerbated by poor intervention description in the primary
literature, the distinction between reinforced and non-reinforced is sometimes unclear. For some
studies, for example, it is difficult to ascertain to what extent the informal caregiver actually received
explicit and structured support, and whether this support constitutes labelling an intervention
“reinforced”.
Of the nine studies included in this review most are RCTs (n=6), with the remainder (n=3) all
assessed using a CCT design. With regard to population, 5 studies included solely patients, and/or
the informal caregivers of patients, who were receiving palliative care due to a diagnosis of advanced
cancer. One study included caregivers of patients with either cancer, COPD or CHF, one study
included caregivers of patients with heart disease only, and two studies included caregivers of
patients with any chronic illness. Four studies were carried out in the US, three in Australia and two in
the UK.
42
Given that substantial heterogeneity was expected among included reinforced home based palliative
care interventions, and that we suspected this heterogeneity could influence effectiveness, we
extracted detailed information with regard to the included populations and interventions. The
characteristics of the nine included studies are given in Appendix 22, which includes the study
design, country of study and participant details.
After we identified the nine included studies, we aimed to group the interventions in a sensible
matter, based on commonalities with regard to theory, design or delivery. To this end, we have
grouped interventions into “Cope interventions”, “Cope-like interventions”, and “others” (See Table
3.2). COPE interventions, focusing on four components – Creativity, Optimism, Planning, and Expert
information, aim to address the specific needs of informal caregivers caring for persons with cancer
at home by helping them develop problem-solving skills (Houts, 1996). Informal caregivers learn to
develop and carry out care addressing both medical and psychosocial problems. Cope-like
interventions share similarities with the COPE interventions described above, but these are not
explicitly grounded in problem-solving theory and thus less uniform with regard to the design.
Services may include needs assessment of informal caregiver needs, training and education of the
informal caregiver in patient assessment, care plan development and social network utilization,
promoting informal caregiver psychological well-being, preparation and support for informal caregiver
in preparing for patient death.
These interventions may have certain similarities with those described above, but not enough to be
considered either COPE or COPE-like. As outlined in Table 3.2, one intervention was organized
around an introduction of various caregiving-relevant themes throughout the time the patient receives
palliative care. The other focuses on the development and utilization of social support networks and
related resources. Both of these interventions provided some form of respite for informal caregivers,
providing some structured time away from the patient.
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Table 3.2 Description of included reinforced home based palliative care interventions
Study ID
COPE Interventions
McMillan 2007 Caregivers received standard hospice care and were additionally taught to assess and manage patient symptoms. The model has four components: Creativity (What could I do to distract Dad from his pain?); Optimism (“I believe I can help Dad with his pain”); Planning (“How can I get Dad ready for our family holiday party?); and Expert information (Where can I go to get help with Dad’s pain?).
McMillan 2013 Caregivers were given the book “Home Care Guide for Advanced Heart Disease”, which outlined the problem-solving approach. A series of home visits (3x) and phone calls (2x after each visit) were used to teach and review the steps in the problem-solving method, using specific patient problems chosen by the caregiver as being highest priority. Patients were also given homework for the time between visits, which was also discussed.
Meyers 2011 Patients and caregivers are provided with the book “Home Care Guide for Cancer”, made up of chapters addressing problems such as physical symptoms, resources, relationships with one’s healthcare team, support or services from family, friends and community organizations. The chapters all follow the same problem-solving formula. Three educational sessions are also given by the trained educator, focusing on addressing a series of patient- or caregiver-identified problems, using the COPE method.
COPE-like Interventions
Hudson 2005 Family caregivers were visited by a trained palliative care nurse two times at home. The first visit consisted of discussing the caregiver role, as well as developing a care plan. In the second visit, previous strategies were evaluated and new strategies were developed. Caregivers were also given a guidebook and audiotape to help them prepare for patient death. In between home visits, a call was made evaluating progress and discussing next steps. Condolences were also sent after patient death.
Hudson 2014 Two groups received a combination of face-to-face visits and phone calls (either 1x and 3x or 2x and 2x) from a Family Caregiver Support Nurse (FCSN). The FCSN assessed needs, prepared a care plan, assisted caregivers in preparing for relative’s death and bereavement, and in referral to other services as required. Caregivers were also given a guidebook focusing on preparation and management of the caregiver role.
McMillan 2007 (support)
A second intervention group in this study received standard hospice care plus supportive visits from the intervention nurse according to the same schedule as the COPE intervention group. The nurse spent time providing individual support to the caregivers, discussing their feelings, fears, and relationships with their patients.
Rabow 2004 On top of a multi-component, comprehensive program directed at the patient, a nurse provided training and support for the informal caregiver through formal classes and informal individual consultation. Patients and their families were invited to monthly support groups that included discussions about symptom management and advanced care planning.
Walsh 2007 Informal caregivers received six visits by trained advisors. Visits consisted of a comprehensive assessment of needs across a wide range of domains (patient care; physical health; need for time away from patient; need to plan for the future; psychological health, relationships and social networks; relationships with health and social service providers; finances), as well as advice, discussion, and emotional support regarding past, present and future issues. If these meetings could not be arranged in person, or if difficult topics were discussed, then telephone could substitute visits.
Other Interventions
Harding 2004 Caregivers took part in weekly 90 minute-sessions where various themes such as welfare benefits, occupational and physical therapy, aromatherapy were introduced. They further participated in group discussions focusing on patient issues and weekly themes. Caregivers could use transportation organized for them to go to the
44
sessions and had someone taking care of the patient at home.
Greene 2012 Caregivers received a minimum of 3 (and potentially more) home visits from the community network facilitator. These visits consisted of comprehensive assessment of caregiver needs, as well as training and support for the caregiving role through a structured booklet and practical advice on care. The CNF helped the caregiver identify his/her own social support network and how this could be utilized in supporting them. The CNF also helped put caregivers in touch with resources they both already knew about, and with new resources as appropriate for specific needs. They also arranged in-home respite to ensure that the caregiver had planned breaks, at least on a weekly basis.
Effects
The intervention effects from the included studies are shown on the harvest plots below in Figure 3.2
and Figure 3.3. Although reinforced models of palliative care focus mostly on the informal caregiver,
some of the included studies assessed patient outcomes, too. Figure 3.2 illustrates that most of the
measures were consistent, showing that reinforced home based palliative care interventions were
neither significantly better nor worse than non-reinforced home based care with respect to patient
outcomes. One study measured patient pain, showing a neutral intervention effect. Five studies
measured symptom control, with a mix of two positive and three neutral effects. Patient QoL was
measured in three studies, all of which showed a neutral effect. Psychological health outcomes were
measured four times across three studies, showing two positive and two neutral effects. Two studies
measured hospitalization, both showing a neutral effect. Two studies yielded three estimates of
patient response, of which all showed neutral intervention effects. One study also assessed patient
satisfaction with care, showing a neutral intervention effect.
Across caregiver outcomes, illustrated by Figure 3.3, reinforced home based models were
inconsistent with regard to effectiveness, with the majority of studies showing no effect. For caregiver
QoL, nine measurements across eight studies yielded a mix of four positive and five neutral effects.
Sixteen estimates for psychological health were taken from eight studies, of which three favoured the
intervention, one favoured the control, and twelve were neutral. Response outcomes for informal
caregivers were measured fourteen times in six studies, yielding four positive and ten neutral effects.
Caregiver satisfaction with care was measured only once, and it showed a neutral effect for the
reinforced home based intervention.
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Figure 3.2 Harvest plots showing intervention effects for patient outcomes
Figure 3.3 Harvest plots showing intervention effects for informal caregiver outcomes
46
Gap analysis and expert consultations
With the completed harvest plots, the review team started the gap analysis. The team thought that
the most interesting and appropriate issues to discuss were; i) the heterogeneity of the evidence, and
ii) why the majority of the effects, as seen in the harvest plots, were neutral with regard to both
patient and informal caregiver outcomes. We identified four main points, which we felt could have
influenced the effectiveness of included interventions or the assessment of effectiveness at the
primary study level. These included the heterogeneity and ambiguity of usual care and non-
reinforced home based care, against which the included interventions were compared; the individual
tailoring based on patient and caregiver needs necessary for many components of palliative care; the
appropriateness of outcomes used in this review, as well as at the primary study level; the study
designs, with which these interventions tend to be evaluated. These aspects, described in Table 3.3,
were used as a flexible structure during one-on-one consultations with four palliative care
practitioners and researchers.
Information stemming from the stakeholder discussions, also outlined in Table 3.3, should not be
taken as “hard evidence”, but rather expert opinion, as it comes from very few individuals and is
based on personal knowledge and experience. Such information can, however, be used to think
critically about the included interventions, especially with regard to the effect, or lack thereof,
portrayed by the harvest plots.
47
Table 3.3 Overview of the discussions within the gap analysis and expert consultations
Identified aspect Gap-analysis Expert consultations Usual care The type of care, against which reinforced care was assessed,
was poorly described in most included studies. Usual care
received by both patients and informal caregivers needs to be
better described to understand why many reinforced palliative
care interventions assessed to date were not effective.
Usual care varies from place to place - not only from country to country,
though there are very substantial differences to be seen at that level, but
also within countries from one location to another. These differences are
present, even if the offered services are technically the same - i.e.
community nurses, with support from specialist nurses and physicians
may be standard care in numerous locations, but what and how much
thereof provided by such services may differ drastically.
Especially the organization of care – who is responsible for what and to
what extent – differs across locations.
The timing of introduction of home based palliative care, both for patients
and informal caregivers, and to what extent this care is integrated with
other aspects of care is highly variant, because guidance regarding the
optimal time for introducing care is lacking, and because this may differ
widely among patients and caregivers.
The support that caregivers receive as part of usual care is extremely
heterogeneous, both between and within countries. Some caregivers
receive structured support throughout the illness trajectory, while others
receive help only when they are overwhelmed by problems and seek care
themselves.
Also the extent to which caregivers are involved in decisions regarding
patient care differs within “usual care”, from virtually none to playing a part
in care-planning discussions.
Tailored care Although some of the included interventions did offer some
flexibility, it could be that for reinforced palliative care to be
effective, targeted and tailored care should be delivered to
those patients and caregivers assessed as needing it.
Care tailored to the individual patient and caregiver, at least to a certain
extent, is seen as the best practice - this could be based on diagnosis,
age, illness trajectory, social surrounding, etc, and the recognition of
such indicators is important.
Being able to assess the needs of patients and/or informal caregivers,
and to inform them of (evidence-based) options is essential.
48
The health and social care professional, however, should not make
assumptions about what patients and/or caregivers need or want, and
they should be involved in these discussions.
It is also important to realize that the needs of caregivers and patients are not static, and will likely change over time and trajectory of the illness. This makes repeated assessments through ongoing communication important.
Assessed outcomes
All of the outcomes assessed in this review have been used in
the primary literature, and are thought to be important for
patients and caregivers. It should be considered, nevertheless,
whether these are most appropriate, and whether certain
additional or alternative outcomes should have been assessed,
both in the primary literature and in this review.
Hard outcomes used in palliative care may only tell part of the story, and
meaningful effects can potentially be hidden among the noise, e.g. in a
population so severely burdened, it may be unrealistic to expect clinically
significant differences in quality of life. It is important, therefore, to ask
patients and caregivers if their care has improved, and what the benefits
of care were.
To select the most appropriate outcomes, it is critical to consider the
goal of the intervention and what it is designed to achieve.
Also outcome importance may differ between subgroups, and it is
important to recognize this when evaluating services.
Study design Included studies encountered a range of problems when
implementing and assessing palliative care services – e.g.
attrition. Study designs, other than those included, may be more
appropriate for assessing the effectiveness of reinforced home
based palliative care services.
Mixed-methods and qualitative research should play a large role in
assessing the effectiveness of services in a meaningful way - it is
important to see what exactly is happening, to hear what patients and
caregivers feel they are getting, as opposed to assuming, based on the
intervention design.
If care is truly based on caregiver/patient assessment, and therefore
truly tailored to the individual, and because the goals of individual
participants will be different, evaluation of care becomes very difficult,
especially in a randomized trial. The idea that the methods used for
assessing effectiveness in palliative care have to be exactly the same
as in other medical fields like pharmaceuticals should be reconsidered
and other study designs – e.g. process evaluations, qualitative studies,
N-of-1 studies – should be considered as well.
49
Discussion of results
The objective of this review was to assess the effectiveness of reinforced home based palliative care
interventions across a range of informal caregiver and patient outcomes. Evidence from the five
studies originally included in Gomes et al’s (2013b) review, as well as the four identified through the
updated searches, graphically summarised in Figures 3.2 and 3.3, show that the observed effects
have been largely inconsistent. A majority of studies show no effect for reinforced home based
palliative care interventions compared to non-reinforced home based interventions. For patient
symptom control and psychological health outcomes, as well as for caregiver QoL, psychological
health and response outcomes, some positive effects were seen, but the overall impact appears to be
neutral for both patient and caregiver outcomes. The gap analysis and the expert consultations,
however, focused on and highlighted reasons, which could possibly explain this ineffectiveness, and
this provides an important complement to the evidence from the primary studies.
The harvest plot, as a method for summarising evidence across studies, is advantageous because it
clearly highlights the “completeness” of the evidence base. It is clear, for example, that patient
outcomes are rarely assessed as part of these assessments. In contrast, informal caregiver outcomes
were the major focus of most of the included studies.
The included studies come solely from developed countries, with four coming from the US. Of the
remaining five studies, three were conducted in Australia and two in the UK. Additionally, the
developed world is not well represented, and the generalisability of these results should be
considered. The UK is, for example, the sole European country represented, and many aspects in, for
example, health system organisation, socio-cultural and socio-economic differences, to name a few,
must be considered when interpreting these results in the context of Europe as a whole. Similarly, the
US and Australia also represent different societies and health systems, organised in different ways,
with different values, and such issues cannot be ignored in this review.
It can be seen from Table 3.4 that study quality varied among the included studies. Although the
Cochrane EPOC risk of bias tool does not provide an overall assessment of the study quality, it
allowed us to rate certain as “Low risk” (green), “High risk” (red) or “Unclear risk” (orange). Six of the
nine studies were RCTs and can thus be considered of higher quality, but the quality among the
RCTs varied as well, while CCTs, due to the non-random intervention allocation, showed an
inherently higher risk of bias (CH). It is possible that we introduced bias into our results through some
of these routes. For example, it was unclear whether most studies sufficiently dealt with incomplete
outcome data. If some studies did not sufficiently deal with this, and if those informal caregivers or
patients lost to follow up were the most severely affected, then the intervention effect may have been
overestimated in these cases. Additionally, blinding of assessment of primary outcomes was only
performed in three studies. If those assessing outcomes were to systematically favour those receiving
the intervention, the effect would be overestimated.
50
Table 3.4 Risk of bias in each study using Cochrane EPOC risk of bias tool
Allo
ca
tion
se
que
nce
ge
ne
ration
Allo
ca
tion
co
nce
alm
en
t
Ba
se
line
ou
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asu
rem
en
ts
Ba
se
line
ch
ara
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ristics
Incom
ple
te
ou
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ta
Blin
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asse
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Ad
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pro
tection
ag
ain
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Re
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g b
ias
Harding 2004
Hudson 2005
McMillan 2007 (COPE)
McMillan 2007 (Support)
Walsh 2007
Rabow 2004
Greene 2012
Hudson 2014
McMillan 2013
Meyers 2011
Furthermore, with regard to bias, it cannot be ruled out that some biases were introduced into the
results through the conduct of the review, although we attempted to maintain to a high level of rigor
throughout. It is possible that our search strategy was not sufficiently relevant or comprehensive with
regard to search terms, or with regard to the number and scope of included electronic databases,
which could result in relevant studies not being identified. Potentially, we could have overlooked
relevant studies at the title/abstract and full text screening stages, although duplicate screening
lessens this risk. It is possible that some data were extracted incorrectly, which could lead to biased
results, but all results were screened in duplicate to minimise this risk. If extracted data were used
incorrectly in creating the harvest plots, the results could be biased. To minimise this risk the harvest
plots were checked multiple times by members of the review team. As described above, the
distinction between reinforced and non-reinforced home based models was sometimes problematic. If
some interventions provided ample support for the informal caregiver, yet described this care vaguely
or poorly in the published paper, it is possible that the list of reinforced services included in this review
understates the actual situation. In reality, support for the caregiver probably exists along a spectrum,
spanning from no support, to some support alongside the patient, to frequent and comprehensive
support, with all interventions fitting somewhere in between. More consultations with experts were
originally planned, but due to scheduling conflicts and a lack of time, only four were possible. It would
have been valuable to have input from more experts, as this would have provided information from a
range of perspectives. The consulted experts were, however, from three different countries and
worked on different aspects of home based palliative care, meaning the information does stem from
different perspectives.
As the results from most included studies show either a positive effect or no effect for reinforced home
based palliative care interventions across patient and caregiver outcomes, it unclear whether such
services will be effective if implemented.
51
The included interventions represent a range of heterogeneous services, and what may prove
effective in one location, setting or context may not be in another. The scarcity and heterogeneous
nature of data identified and included in the review did not allow for meta-analysis or for detailed
subgroup analyses, meaning aspects such as active intervention ingredients, optimal intervention
timing and duration, and most appropriate target population, among others, are still unclear. The
expert consultations did, however, shed some light on potentially important and relevant implications
for practice and research. A clear understanding of usual care, i.e. of what type of support patients
and caregivers already receive, may be important for identifying what additional, alternative or
complementary services could be most effective. Assessing patient and caregiver needs frequently,
and tailoring care based upon what care, how much thereof, and when the care is needed may lead
to more effective interventions. The outcomes used to assess reinforced home based palliative care
interventions should also be revisited, and hard outcomes such as quality of life, improvement in
psychological health outcomes, etc. should possibly be supplemented with more qualitative
outcomes. It is possible that traditional outcomes are not sensitive enough in this highly burdened
population, and qualitative outcomes can help evaluate whether and to what extent patients and
caregivers feel interventions, as well as certain intervention components, are helpful and effective.
Researchers should also consider what study designs may be most appropriate for answering
questions of effectiveness, and may look to designs other than the RCT. Process evaluations,
qualitative methods, and N-of-1 studies, for example, are all designs that could prove useful for
answering particular questions regarding intervention effectiveness, including active intervention
ingredients, optimal intervention timing and duration, and most appropriate target population.
Reflections
We reflected on issues of complexity, integration, stakeholder involvement and the value and
application of the methods developed for effectiveness HTA.
Complexity
It was because of the complexity related to reinforced home based palliative care interventions, i.e.
multiple components, multiple delivery agents, multiple recipients, and numerous important outcomes,
that we decided to forego meta-analysis, presenting our findings instead using the harvest plot,
complemented with the expert consultations. The variety of populations studied, the wide range of
intervention assessed, and the numerous outcomes of interest did complicate traditional systematic
review methods, and led to a large search strategy, the necessity of very detailed data extraction and
the management of large amounts of information. This does not have to be viewed negatively,
however, as the harvest plots do allow for the efficient handling and clear presentation of
heterogeneous outcome data.
Integration
Integration was especially important in informing scope of the review. The definition of key aspects,
including population, intervention and outcomes of interest, were greatly influenced by the logic model
for palliative care developed within INTEGRATE-HTA. This logic model was informed by the literature
and by stakeholder input, and received input from a range of colleagues with various expertise, e.g.
relating to ethics, economics, and socio-cultural. Consequently, we feel that aspects of the review,
such as the selected populations, interventions and outcomes, reflect the holistic nature of palliative
care.
52
Ideally, this effectiveness assessment, as well as all effectiveness assessments looking at complex
interventions, could be integrated further at various stages of the review process. Post-hoc subgroup
analyses, for example, could be performed based on input from socio-cultural or economics experts,
in order to highlight interesting relationships possibly not in the scope of the current review.
Similarly, the work on patient preferences and treatment moderators could have informed potentially
interesting subgroup analyses, and such work would be appropriate in future research.
Stakeholder input
The additional workload incurred by the review team due to the involvement of expert stakeholders,
which included the gap-analysis and expert consultations, as well as working the resulting information
into the effectiveness assessment post-hoc was not substantial, and can be considered minor
compared to the information gained from this process. For the respective experts who we consulted,
the workload was also light, consisting of approximately 30 minutes of preparatory work, and 60
minutes for the discussion, an important factor given the busy schedules of experts in this field.
Experts also seemed generally happy to dedicate this time to share their experience related to
effectiveness of interventions and the conduct of palliative care research (see Appendix 24 for a
summary of stakeholder feedback on their involvement in the effectiveness analysis). They also
commented that they found the presentation of evidence using the harvest plots a useful and clear
way of organizing the complex evidence.
Although the information stemming from the expert consultations is based only on the thoughts and
experience of the respective palliative care experts, and should therefore, not be taken as hard
evidence, some very interesting points, within which to frame our review results, did arise. We
discussed methodological and clinical aspects that may have hindered detecting an effect of
reinforced home based palliative care, as well as those that may have limited effectiveness of the
included interventions. As our initial assessment showed evidence only of limited effectiveness, such
information offered a valuable complement for researchers and clinicians looking to develop,
implement and evaluated similar interventions in the future.
Value and application of methods
The use of the harvest plot for evidence synthesis in this review, facilitated an efficient handling of a
very heterogeneous evidence base, and could also be applied in other reviews of complex
technologies. Similarly, the involvement of stakeholders in processing the evidence allowed us to
further explore the effectiveness of included interventions, and could be relevant for other complex
technologies, where many relevant questions may still remain after the initial assessment.
Conclusion regarding effectiveness
The harvest plots indicate that much of the evidence showed such interventions to be ineffective, with
some positive intervention effects, and very few negative intervention effects. This review emphasizes
even further that home based palliative care interventions, including both reinforced and non-
reinforced models, are complex due to a range of multifaceted tailored services, multiple stakeholders
and outcomes. Consultations with researchers and practitioners working in the field of home based
palliative care allowed us to further explore and discuss the results and implications of these results
with those who have experience and knowledge in the field, providing fresh perspectives on the
evidence and its implications for practice and further research.
53
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54
The economic aspects of home and reinforced home based palliative care
Background
This section reports an economic assessment of reinforced models of home palliative care that is
home care models which include a component of intentional or explicit carer support, compared with
non-reinforced models of home palliative care. In describing the cost and cost impact of an
intervention it is necessary to have a detailed and specific description of both the intervention and the
system within which it is to be implemented. In light of this it was decided at an early stage to confine
the scope of the economic element of this demonstration case study to be UK specific. A common
criterion in making decisions about the scope of the model was to make the economic evaluation
sufficiently specific to enable the estimation of costs/resource impacts but sufficiently transparent and
generic to maximise its relevance and to enable readers to translate findings to their own
environment.
Aims
To test and refine the economic guidance produced in the INTEGRATE-HTA project and to assess
the economic impact of reinforced models of home palliative care.
Methods
The economic assessment is based on the development of a model of reinforced home palliative
care. The model is developed according to the process outlined by the guidance for economic
modelling of complex interventions in complex settings (Chilcott et al, 2015). Evidence informing the
model is drawn from a review of economic evidence relating to reinforced home palliative care, and
judgements elicited during an economic stakeholder workshop and series of telephone based
interviews. The process, evidence and derived model are described in this report.
Process for development of the health economic model
Model development follows the five phase process outlined in the guidance for using a systems
approach for economic modelling of complex interventions in complex settings (Chilcott et al, 2015)
'Guidance for assessing effectiveness, economic aspects, ethical aspects, socio-cultural aspects and
legal aspects in complex technologies'}. The five phases progress with a degree of iteration and are:
understanding the decision problem, problem oriented conceptual modelling, design oriented
conceptual modelling, model Implementation and checking and engaging with the decision problem.
This process is managed with an explicit phase of aligning the modelling process with the decision
problem. These phases are described in more detail below.
Understanding the decision problem
The first stage in the modelling exercise is to develop an explicit understanding of the decision
problem that captures the views and perspectives of different stakeholders. This phase is involved
with describing who will use the outputs of the modelling, the economic question to be addressed and
should seek to identify specific interventions for assessment.
55
The ‘understanding of the problem’ identifies:
The economic research question
Decision making context
Definition of the stakeholders and roles
PICO – defining the Population, Intervention, Comparator and Outcomes
Underlying intervention theories
This phase is concerned with; describing factors thought to be relevant to the problem, developing a
conceptual model of hypothesised causal chains, identifying potential relationships between actions
and outcomes and identifying interventions for assessment. This description needs to capture the
breadth of problem understanding of stakeholders supported by initial explorations of the available
empirical evidence. This phase identifies the perspective and key outcomes of interest for each of the
stakeholders, that is the consumers (e.g. patients), actors (e.g. healthcare professionals) and problem
owners (e.g. healthcare commissioners).
Problem oriented conceptual modelling
The aim of the problem oriented conceptual modelling phase is to develop descriptions of the health
systems that enable the potential impact of the intervention on economically relevant outcomes to be
made explicit. In addition it enables judgments to be made about the design of a model to produce
quantitative estimates of these outcomes.
Two conceptual models should be described:
1. The disease logic model: identifying the explicit value proposition for intervention, that is to
identify the theory underpinning the intervention and identify how the intervention is thought to
impact on health and wellbeing outcomes of economic relevance. This case study uses a
cognitive mapping approach for representing the disease logic model.
2. The activity/resource pathway model: A descriptive model of the activities, services and
resources, identifying actors involved in the delivery of the intervention and the system
impacted on by the intervention. Current service provision will be described. The specific
purpose of this model is to provide a basis for justifying the resources and costs included in
the design oriented model structuring phase.
Design oriented conceptual modelling
The specification and design of the quantitative economic model. This phase involves iteration with
the development of the problem oriented conceptual models and further information gathering
exercises, including:
Definition of the type of model (for example Markov, Decision tree, analytical, ABS),
A visual diagram of the model appropriate to the above type,
Specification of the functional relationships and parameters forming the model,
Description of approach to parameterisation (for example calibration, synthesis or both) and
Specification of data sources.
56
Model Implementation and checking
A quantitative model is developed according to the design set down in the design oriented conceptual
modelling phase. The report of the elicitation exercise gives a detailed account of the discussions
underpinning this phase.
Engaging with the decision problem
As a demonstration case study the project is constrained by the absence of a specific real-world
decision context for the economic evaluation. Rather, the focus of the assessment was driven by the
contribution of stakeholders through the early SAP process. The relevance and definition of the
economic outcomes for decision makers was discussed in the stakeholder sessions.
Aligning the modelling process with the decision problem
The model development process describes a generic and scalable approach to economic model
building and analysis. At the start of each project there is a need to adjust and define the approach for
the context of the specific decision problem. This is to ensure that the economic modelling exercise
meets the project aims within resource and time constraints but also in the light of other political,
contextual and environmental constraints.
The project constraints included:
Time - The timescales of the INTEGRATE-HTA project and early scoping work meant that the
economic evaluation needed to be completed between November 2014 to May 2015.
Resource - The INTEGRATE-HTA project supported two health economic modellers (part
time), in addition to information resources, project coordinator, an administrative assistant and
other members of the wider European INTEGRATE-HTA team contributed to the economic
case study. There was also significant contribution from palliative care experts and Public
Patient Involvement (PPI).
Political, context and environmental - The disaggregated nature of the local palliative care
system means that there are a high number of potentially relevant stakeholder organisations,
with a consequent challenge in coordinating, managing and administrating the project process,
and particularly stakeholder engagement. At a wider level palliative care is the subject of a
number of national initiatives and reforms including a national enquiry and palliative care
funding review and associated pilots, with an attendant high level of public attention. This has
a number of impacts including potentially reduced inertia in the system with a greater
readiness to embrace change, rapidly changing definitions and terminology and the
emergence of new research and evidence. The challenge was to undertake casework with
local relevance sufficient to justify stakeholders in the contributing time, yet is sufficiently
generalisable to have relevance beyond the local palliative care system, i.e. nationally and
internationally.
In this project developing the understanding of the decision problem and the problem oriented
conceptual modelling were driven by:
a) Inputs from the case study expert panel and the early stakeholder consultation, reported
elsewhere (See chapter 2)
b) A review of reviews in palliative care, reported elsewhere (see chapter 2)
c) A review of the economics literature in palliative home care, reported fully in Appendix 23A.
57
An existing systematic review by Gomes et al in 2013 reported on the effectiveness and cost
effectiveness of home care and identified 23 studies in total. (Gomes et al 2013) Four studies
were identified as reinforced home care studies.
The review of effectiveness evidence updated the systematic review by Gomes et al 2013
(Burns et al, 2016). A review of the economic evidence contained in these newly identified
studies was undertaken. Individual papers were reviewed in order to gain a more in-depth
understanding of the interventions and comparators and the key issues identified in relation to
potential resource implications and cost drivers within the palliative care system.
d) A stakeholder consultation process
To facilitate economic stakeholder engagement the original protocol specified: 1) a
stakeholder workshop at the start the process in order to inform the understanding of the
problem and derive information for the conceptual modelling and 2) a second workshop at the
end of the process to explore the feasibility of filling data gaps in the economic model and
present results for discussion, there was also provision for 3) an elicitation exercise for
identification of model parameters where elicitation of subjective judgement is: a) feasible and
b) worthwhile.
Stakeholders for the economic evaluation were identified from the group of professionals and
lay people who were advisors to the case study and academics and practitioners with an
interest in palliative home care or reinforcement interventions. The selection of economic
stakeholders was constrained by the need to spread the stakeholder workload across the
different aspects of the case study, e.g. effectiveness, ethics, socio-cultural context, and the
limits on project timescales and resources. Nine people agreed to contribute as economics
stakeholders. The advisory group comprised seven females and 2 males, where a religion was
specified this was either atheist or Christian and the group where predominantly white with
one black British member. The stakeholders included a lay person with experience of palliative
care, academic palliative care practitioners including nursing, consultants and Professor
grades and palliative care service managers from the charitably funded hospice sector.
Results
The results section presents the results of the key activities undertaken in implementing the five
phases of the model development process.
Firstly the review of the economics literature in palliative home care and a stakeholder workshop
which informed the understanding of the decision problem (phase 1) and the problem orientated
conceptual modelling (phase 2) is reported. Following the stakeholder workshop, the conceptual
modelling phase, including the problem orientated and design orientated conceptual modelling
process was completed (phase 2 and phase 3). The limitations of the published evidence base were
identified and the results of the expert elicitation exercise to provide input parameters to allow model
implementation (phase 4) are reported. The draft model was developed and initial analyses
undertaken. Results of the elicitation process and the draft model results were presented back to the
stakeholders for validation purposes and to allow initial discussion of how these results would be used
in the decision making process (Phase 5). The final economic modelling results are presented below.
Review of the economics of reinforced carer support and home care models
A summary of the review of the economic evidence is given below. Full details are provided in
Appendix 25.
58
As part of the review of effectiveness evidence the systematic review by Gomes et al (Gomes et al,
2013) was updated to identify new studies of reinforced home based palliative care and usual (non-
reinforced) home based palliative care (Burns et al, 2016). A review of the economic evidence
contained in these newly identified studies is reported below.
In addition, all papers of either reinforced home care or home care which reported total costs (as
identified by Gomes et al and the updated searches) were reviewed. An analysis of total costs is
important in order to gain a more in-depth understanding of the key issues in relation to the potential
system-wide resource implications of a new intervention and the cost drivers within the entire
palliative care system.
The original systematic review by Gomes et al identified 23 studies – 19 home care studies and four
reinforced home care studies. The authors reported there was no economic evidence contained in the
four reinforced home care studies. The updated searches identified 10 additional studies – six home
care studies and four reinforced home care studies. (Burns et al, 2016) No economic evidence was
reported in the four newly identified reinforced home care studies.
Of the 19 home care studies identified by Gomes et al, six considered the impact on total costs of
home palliative as opposed to usual care, alongside the evaluation of clinical effectiveness. (Brumley
et al, 2007, Greer et al, 2006, Higginson et al 2009 , Hughes et al 1992, Tramarin et al 1992, Zimmer
et al, 1985) All were reported by Gomes et al to be high quality studies. Five of these studies were
Randomised Controlled Trials (RCTs), one was a controlled before and after study. One of the six
studies, Higginson et al, was UK based. The generalisability of the results of other studies to the UK is
unclear. (Gomes et al, 2013). None of the six studies identified by the updates searches reported total
costs. Therefore, in total, six of the home care studies were identified as reporting total costs.
All six of these home care studies reported lower costs in the intervention groups, although
differences were statistically significant only in the study by Brumley et al 2007.(Gomes et al,2013) In
this United States (US) –based study, 297 people with cancer, cardiac heart failure (CHF) and
chronic obstructive pulmonary disease (COPD) with a prognosis of 1 year or less to live, were
randomised to usual home care (following Medicare guidelines for home healthcare criteria) or usual
care plus in-home palliative care - an interdisciplinary home based palliative care programme aimed
at improving patient’s quality of life. The intervention was offered until death or transfer to a hospice
program and was more effective than usual home care in some outcomes, for instance patients were
more likely to die at home and expressed a greater satisfaction with care and equally effective in
others, whilst also saving costs. Cost savings were the result of patients being less likely to visit the
emergency department (p<0.01) or be admitted to the hospital (P<0.001) In the UK-based study by
Higginson 2009 (50 people with Multiple Sclerosis and their caregivers) of early referral to a palliative
care team for assessment and short term support, the intervention was more effective in some
outcomes, e.g. reduction in caregiver burden and equally effective in others, but the study reported no
difference in costs. In the remaining four studies there were positive, null and negative clinical
effectiveness results and the statistical significance of differences in outcomes was not reported.
The studies covered different disease areas, including cancer, AIDS and multiple sclerosis. Most
interventions involved use of a multidisciplinary team, typically offering assessment and individually
planned care with home visits; however the mix of health professional input to the teams varied
across studies. Volunteers were included in some teams and not in others. Variation between the
size and skill mix of the team and the nature of the services offered will impact on the cost of the
intervention, but also the potential impact it will have on patients and carers and the rest of the
palliative care system. Timing of the intervention also differed between studies. For instance in
Higginson et al the intervention comprised an early specialist palliative care intervention at the time of
diagnosis, whilst in other studies support was targeted closer to the end of life (EoL).
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In some cases the intervention was for a limited period e.g. in Higginson patients had one to three
contacts (visits/phone calls) only; in other studies support was on-going. In addition, the comparators
varied between studies and were typically poorly described.
In some case the comparator was standard hospital care e.g. Tramarin et al 1992, and in others it
was usual or standard community/home care. It is important explore how these differences might
contribute to the individual findings rather than to try to attempt to draw conclusions based on
aggregation of the results of these individual studies.
The six studies report a wide range of outcomes – different outcomes in different studies, as well as
multiple outcomes within same study. This makes it difficult to compare results across studies.
Outcomes of both patients and carers will be important for considering the effectiveness of reinforced
carer support interventions, as well as economic implications resulting from changes in usage of other
palliative care services. The impact on the rest of the health care system is rarely considered within
these studies, e.g. the impact on informal caregivers, hospice usage, and usage of other support
services. Quality of life (QoL) outcomes are reported using many different tools and rarely in a form
that allows the cost per quality adjusted life year (QALY) to be reported. It is unclear from the
research presented what the most relevant outcomes for economic decision making are in this
context and further consideration of this issue is needed.
In addition, the period of analysis varies between studies. The impact of the intervention on resource
use/costs and outcomes will be influenced by how long the intervention is offered and at what stage of
in the palliative care phase, e.g. an intervention focussed on the last 2 weeks would be expected to
report very different outcomes to one considering the last 12 months. Timing of the outcome
measurement is also relevant - whether outcomes were measures during and/or post intervention.
Therefore many of the studies are not directly comparable.
The perspective used for the analysis in the six studies is typically narrow – often focussing on health
care costs only, rather than the broader societal perspective recommended by Preston et al. (Preston
et al, 2012) Typically direct costs only are considered.
In conclusion, the small number of reinforced home care models identified have reported limited
economic evidence; none have reported total costs. A review of usual (non-reinforced) home care
models reporting total costs suggests that the economic evidence to date is inconclusive. A wide
variety of effectiveness outcomes are reported and these are typically mixed – with some positive,
some negative and some neutral. Costs in the intervention groups of the non-reinforced home care
studies are often lower than in the comparator arm, although differences were statistically significant
only in the study by Brumley et al 2007. In this study, lower costs in the intervention group resulted
from reduced numbers of emergency department visits and hospital admissions for patients in the
intervention arm. Differences between the six studies are readily apparent and may have contributed
to differences in the findings. Some studies reported cost saving “in some circumstances”, e.g.
Tramarin et al 1992 reported cost saving if strict criteria were applied to patients receiving the service.
Studies need to provide more detail in relation to the exact nature of the intervention and the
comparator, and seek to answer the question “Under what circumstances might the intervention be
cost effective?” rather than just “Is the intervention cost effective – yes or no?” It is unclear what
conditions are required to ensure that an intervention would work if transferred to another setting, e.g.
is a primary caregiver required and what features of baseline services or the intervention are
important to ensure the intervention will work within the existing health care system?
The findings from this economic review contributed to the preparation of the briefing document for the
stakeholder workshop.
60
Summary of stakeholder workshop
A stakeholder workshop was held on 5 February 2015 to inform the understanding of the decision
problem and derive additional information for the conceptual modelling phase of the project. A brief
summary of key issues raised during the meeting is presented below. A more detailed description of
the stakeholders involved is given in Appendix 26
The definition of (non-reinforced) home palliative care models, as “experienced home care teams of
health professionals specialised in the control of a wide range of problems associated with advanced
illness - including physical, psychological, social and spiritual aspects “ was discussed and agreed by
the stakeholders. The home palliative care model was described by stakeholders as a “nexus of
systems around a patient”. A wide range of service models exist in practice, across a spectrum
ranging from comprehensive (e.g. West Cumbria Hospice at Home, Midhurst MacMillan Specialist
Palliative Care Service ) to disaggregated models with many providers of services all contributing to
the care of the patient. A common set of roles exist, but with a high variation geographically in how
those roles are fulfilled by the network of services. The roles are listed in Appendix 26. Key issues
raised in discussion included:
Current lack of clarity exists on who is /should be responsible for the patient’s care and on
ultimate responsibility for assessment of needs of the patient/carer, for example relating to
equipment, help with meals etc.
Strong commissioning is needed with clearly defined pathways, giving clear indication of who
is ultimately responsible for the patient. Good communication and coordination is vital to
ensure carers’ needs are assessed and met by a variety of services.
Removal of medical paternalism in recent years & the move towards putting the patient in
control may not always be what the patient wants, needs or is capable of and is potentially
stressful.
Key to developing an effective palliative care system is to develop networks in collaboration
(funding access /commissioning pathways). Comprehensive service models, such as West
Cumbria Hospice at Home, enable such collaboration. In a disaggregated system an
effectively functioning multi-disciplinary team (MDT), which enables continuous decision
making, and reaches across funding boundaries, is vital.
Carers are likely to receive some informal support from members of the home care team
under current non-reinforced home care services e.g. support and advice about symptom
management and what to expect as the disease progresses. Need to be very clear about what
makes a reinforced home palliative care intervention distinct from conventional home palliative
care.
The definition of reinforced home palliative care models as “existing home palliative care with an
additional component of caregiver support” was discussed and accepted. However they indicated
“reinforced” was not a commonly used term. The stakeholders recognised reinforced palliative home
care models as a vitally important issue and supported the research question. Key issues raised
included:
Stakeholders interpreted the definition of the intervention more broadly than the focus of
psycho-educational support as implied by the Gomes et al systematic review (Gomes et al,
2013), and specifically discussed a wider range of carer support needs including such as
practical skills (including medication delivery), ‘after caring’ preparation and symptom
identification
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Practical implantation issues should be anticipated e.g. .difficulty in releasing carers for
training (“Uptake high but Do Not Attends higher”) - therefore an effective sitting service needs
to be part of the intervention.
An additional benefit/opportunity of formal carer training might be the development of
qualifications and/or certification that recognises valuable carer skills and assist with the
development of a future beyond caring.
Important for project to assess ”If carer support can make a valuable contribution?”. Prime
issue is “not just does it work or not – but when does it work?” Also important to understand if
the impact of intervention can be generalised into the stakeholders specific setting
A discussion of the stakeholders within the palliative care system associated with reinforced home
palliative care and their goals raised a number of issues:
The potential move towards personalised health budgets might make the patient/carer an
“owner” of the system.
The appropriate commissioner for the new intervention would depend on where the reinforced
carer support service sat in the wider palliative care system and who it is targeted at.
The timing of the intervention was considered highly important for effectiveness. It would be
optimal when the carer is in the best position to acquire and make best use of the information,
training and support - this would probably be associated with early referral.
Due to the system complexity the intervention needs to recognise and enable multiple referral
pathways, which would require strong commissioning.
Goal of patients health & wellbeing - 100s of potential outcomes are available with little current
agreement on what to measure. One particular outcome set which may be gaining acceptance
for practice nationally is the Outcome Assessment and Complexity Collaborative (OACC) suite
of measures
Carers goals - overall goals for carers need to recognise both the positive and negative
impacts of caring.
Goals of health & social care professionals and decision makers are less clearly defined - in
addition to health, wellbeing and resource objectives, other goals may exist, for example
public perception of the service may be important,
Funding of palliative care is a very complex, highly dynamic context - and is therefore
England specific. Even within England there is a high degree of variability in how palliative
care systems are funded.
Although the proposed scope and the problem orientated conceptual model formed part of the briefing
document sent out prior to the stakeholder workshop there was insufficient time to discuss these
issues in any depth during the meeting.
The problem oriented conceptual models
The health and wellbeing model seeks to clarify the potential causal chain between the reinforcement
interventions, their immediate impact and the overall goals and outcomes of the different stakeholders
including patients and carers, health professionals and decision makers.
Figure 3.4 identifies how the reinforcement interventions are thought to make a positive impact on
carers and patients and contribute to the achievement of goals. The figure is based upon the EAPC
white paper on improving support for family carers (EAPC Task Force on Family Carers, 2010,
Steinhauser, 2000), previous discussion on carer and patient outcomes and the description of the
reinforcement interventions presented in the review by Gomes (2013). This conceptual model was
presented to stakeholders for validation and discussion in the 5 February workshop briefing paper.
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Figure 3.4 Reinforced home palliative care intervention theory
A description of the key activity/resources is given below. Models of home care and reinforced home
care will potentially incur costs to the health and social care services and broader societal costs,
including costs to the patient and their caregivers.
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Health service and social service perspective
The nature of the services offered and resource consumed by the palliative home care service
(comparator arm) will vary according to the exact nature of the baseline services provided. Based on
a review of the studies in Gomes et al 2013 and discussions during the stakeholder workshop the key
potential staffing resources relating to specialist/intermediate palliative home care teams and
generalist PC services are listed below.
Nurses – Specialist Palliative Care, generalist, community matrons, district nurses, nurse
practitioner.
Doctors- Consultant – PC specialist, geriatrician and disease specific, General Practitioner.
Others - social worker, social care assistant, health care assistant, physiotherapist,
occupational therapist, dietician, priest/chaplain/pastoral worker, psychologist/ psychiatrist ,
counsellors, pharmacist (hospital/community) , secretary /administrators, bereavement
coordinator, carers/ companions, volunteers
Other non-staff costs may include travel, equipment and overheads.
The addition of a reinforced carer support intervention may require the use of additional resources
(e.g. recruitment of new staff to run the service) or may be implemented by changing the work
patterns of existing staff. Increased workload is likely to result from involvement in providing formal
training to carers, requiring additional scheduled visits with carers. Decreased resource use may
result from reduced out of hours/emergency visits and avoided hospital admissions and reduce GP
and nurse visits , if carers are better able to manage the care of the patient and avoid crisis situations
from developing and potentially, reduced need for bereavement support if carer quality of life is
higher.
Societal perspective
A societal perspective is potentially relevant for this analysis. Taking into account a broader societal
perspective would allow additional resource use/costs to be considered. A framework for the
assessment of such costs is provided by Guerriere and Coyte (2011). This includes:
- Out of pocket expenses (patients and carers) e.g. travel, equipment, privately financed home
care givers
- Caregiver time losses (time lost from paid employment and household/leisure activities)
- Employer – time missed by employee
- Insurance (in countries ,where applicable)
However lack of evidence and limited project time/resources resulted in only the narrower health
service and social service perspective being considered.
Aspects of complexity
Table 3.5 describes some of the aspects of complexity which are relevant to considerations of the
economics (and to all of the assessments undertaken – i.e. ethics, legal, sociocultural etc.) of
reinforced home palliative care arising from the literature and stakeholder engagements.
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Table 3.5 Aspects of complexity relevant to considerations of the economics of reinforced home Palliative care
Characteristic Application to palliative care
1. Multiple perspectives
The economic stakeholders identified include:
Customers: Patients, carers and families. Though palliative care engages with customers holistically, each individual will have different perspectives reflected in their goals.
Actors: Health and social care professionals, people working in the charitable and voluntary sectors.
Owners: Local and national health and social care managers and policy makers, charitable and voluntary sectors agencies managers and policy makers.
All stakeholders have differing perspectives related to goals and outcomes.
2. Indeterminate phenomena
The philosophy of individualised palliative care that responds to patient and caregiver preferences means that interventions should be flexible and tailored to individual need. Patient and carer needs will change over time, especially if palliative care is provided at the end of life meaning that palliative care has to be flexible to changing circumstances. Furthermore, the concept of palliative care has evolved over time, initially being associated with cancer and more recently evolving to be appropriate for patients with any life-threatening or life-limiting illness.
Home based palliative care is not one single clearly defined/delimited intervention; it has been described as a “nexus of services around a patient”. Further there is not a clear definition of the target population in terms of a) underlying disease, b) functional status, capability or need and c) time of referral within the disease trajectory, i.e. early or late.
Similarly reinforced carer support is not a single well defined intervention and may contain elements of support that are routinely provided within conventional care (e.g. informal training/education of carers).
Service goals are indeterminate: stakeholders discussed equity (reinforced carer support interventions should be offered to all) vs efficiency (interventions targeted at those in greatest need or with the greatest potential to benefit).
3. Uncertain causality
The COPE intervention within the set of reinforced home palliative care interventions was unique in having an explicit underpinning theory. However complexity of context makes the interpretation of empirical evidence base difficult, even well designed trials have difficulty determining causality.
Care providers from a range of agencies work with patients who have many different diseases and illness trajectories any combination of which may interact differently with the causal chain of the intervention. For example the COPE intervention whilst demonstrating some effects in cancer patients, found no effect when trialled in chronic CHF patients. Qualitative research suggested this may be due to the longer disease natural history meaning that patients and carers may have already developed coping strategies and therefore there was a lesser potential impact of COPE towards the end of life.
Additional difficulties for evaluation include ethical concerns about manipulating interventions considered to be beneficial to patients along with pragmatic problems of recruitment, attrition, data collection and missing data {Grande 2011}(McMillan 2003).
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4. Unpredictable outcomes
Stiel et al’s (2012) review identified over 500 outcome measures being used in palliative care. Hence, there is often a lack of consistency with regard to which outcomes are measured and the tools used to do this.
Although there is increasing convergence in terms of outcome measurement tools, (e.g. OACC suite, AKPS, VOC, Zarit and IPOS) outcomes continue to cover a broad range of domains.
Furthermore there is a great distance between the set of outcomes discussed and the single utility outcomes preferred by health economists to support traditional resource allocation decision making.
Additionally, there is uncertainty about which outcomes are most appropriate for the wide range of stakeholders, and how to balance these, especially if they are conflicting.
5. Historicity, time and path dependence
The practice of palliative care has changed over time and the philosophy of individualised care limits the generalizability and repeatability of an intervention.
The palliative care context within which the reinforced carer support intervention might act is dynamic. For example, stakeholders mentioned a number of on-going movements in the health and palliative care system that would impact on intervention effectiveness in England. The introduction of the ‘GP Contract’ in 2003/4 removed GPs 24hr responsibility for the patient, the move towards District Nurses providing a task based service and moves towards the removal of medical paternalism and ‘putting the patient in control’ would all impact on the coherence of the care system. Palliative care funding structures have a major impact on the provision of services and have been and remain subject to ongoing reform. These interconnected dynamics would all potentially moderate the intervention impacts for instance by affecting the ability of carers to divert patients from avoidable admissions to hospital or impact on the level of nursing care required.
The Liverpool Care Pathway (LCP) demonstrated an extreme example of path
dependence. In the UK, even though the LCP initiative had operated effectively in a
community setting, difficulties experienced in the acute sector meant that the
initiative would not be repeatable in the UK. This aspect of complexity is further
demonstrated by the fact that although the LCP has been abandoned in the UK, it
has been adopted in other countries across Europe with a different historical
pathway.
Further controversy over the LCP, an associated enquiry and press attention,
together with the recent Palliative Care Funding Review had “galvanised attention”
on palliative care services which would necessarily affect the potential for change in
the system.
Note the extent of these dynamic aspects would vary even throughout the UK and internationally the palliative care systems would potentially commence from very different starting points.
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Scope for economic evaluation
The scope of the INTEGRATE-HTA project focused on reinforced carer support interventions. As
discussed by our stakeholders this encompasses a broadly set of interventions, with varying modes of
action, costs and impacts. For the purposes of the economic analysis a more specifically defined
intervention was required. Based on the stakeholder workshop (reported above) and a follow up
teleconference with local stakeholders the COPE intervention was selected to be the focus of the
economic modelling (McMillan et al, 2007).
COPE is a brief pyscho-educational intervention. It is a 9 day intervention, typically with 1 to 3 visits
and phone calls in-between. It is theoretically grounded (drawn on stress process models and
problem-solving training and therapy) and aims to teach problem solving methods to carers to assess
and manage patient symptoms.
The COPE intervention was selected on the basis that:
It is a theoretically- grounded intervention
It offered the most comprehensive evidence on effectiveness. The impact on both carer
outcomes and patient outcomes have been reported. The intervention has been applied in two
different groups of patients - cancer patients (McMillan et al, 2007) and in heart failure patients
(McMillan et al, 2013).
Detailed information on the intervention was available from the intervention manual (personal
communication McMillan); this facilitated costing of the intervention and discussion of the
potential impact during the elicitation exercise.
The final scope for the economic analysis is given in Table 3.6 below:
Table 3.6 Final scope for economic evaluation
Population Patients (adults aged 18 years old and over) and their carers
Intervention Reinforced models of home palliative care defined as existing home palliative care with an additional component of caregiver support. The reinforced model includes a specific programme designed to support lay / family caregivers – the COPE intervention.
COPE is a brief pyscho-educational intervention - a 9 day intervention, assumed to comprise 3 visits to carer and phone calls in-between.
Comparator Home palliative care (non-reinforced) defined as a service ‘providing comprehensive specialist or intermediate palliative/hospice care aimed at different physical and psychosocial components of palliative care in which it was clear the majority of service contacts were established while the patients were at home’. Carers may receive some informal support from members of the home care team, for instance support, and advice about symptom management and what to expect as the disease progresses.
Outcomes Health and wellbeing outcomes for patient and carers (As defined in the review of effectiveness evidence) Economic and resources outcomes for decision makers and health and social care professionals
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The design oriented conceptual model
Following the stakeholder workshop and the completion of the conceptual modelling phase, the
specification and design of the quantitative economic model was considered.
Scope of the economic model: Intervention - The COPE intervention was selected to be the focus
of the economic modelling (McMillan et al, 2007). There are four key components to the training:
Creativity – thinking about problems from different perspectives, Optimism – e.g. I believe pain can be
controlled, Planning – setting sensible goals and planning how to achieve them, Expert information –
what they can do to help the patients and when they need to get expert help and how. Caregivers are
trained to use assessment scales for pain, dyspnoea and constipation. In the COPE trials caregivers
received training at home, but not in the presence of the patient; a sitting service was therefore
provided.
Choosing the model structure – The economic guidance provides a series of questions for aiding
the choice of model structure depending on data availability, accessibility of any existing relevant
good quality economic evaluation and the expertise of the modeller. (Chilcott et al, 2015). In this case
study the primary constraint determining the model structure and role is the availability and quality of
evidence.
Not only is there a lack of direct evidence on economic and resource consequences of the
interventions, but there is a general lack of robust evidence regarding the broader palliative care
systems in which the reinforced carer support interventions may act, exacerbated by the complexity of
the system. In particular there is little direct quantitative evidence demonstrating causality and
allowing a scaling of relationships. Thus the role of the economic modelling in this case study is
exploratory rather than definitive. It is not possible from the currently available evidence to determine
the cost effectiveness sufficiently well to support simple commission/don’t commission economic
decision making concerning the COPE intervention (and more broadly the range of reinforced carer
support interventions). The role of the economic modelling is therefore restricted to prioritising and
planning further evaluations of reinforced carer support, or to aiding in the design and modification of
reinforced carer support interventions.
At the outset of the work there was no intention to use the reinforced carer support economic model
for other purposes beyond the INTEGRATE-HTA work; there were therefore no external requirement
to consider in determining model structure. As a result, the model uses the simplest possible
structure, and tabulates potential first order resource impacts across the range of services involved in
the delivery of the palliative care service.
The model boundary – The scope of the model is primarily on the public health and social care
services, but patient and carer impacts are also considered, where evidence allowed.
The stakeholder discussion identified a list of the resource types engaged in the provision of palliative
care services, this resource list was reviewed by all stakeholders to identify those resources
potentially impacted on by the reinforced carer support either directly or indirectly. Those resources
where a potential impact was suggested were included in the analysis and formed the model
boundary. Further details are included in the reporting of the elicitation (see Appendix 27).
The model did not include carer costs due to lack of evidence. However it is widely acknowledged that
the economic costs and implications of providing palliative care to a family member are likely to be
significant at household level. A recent review by Gardiner et al 2014 explored the financial costs and
impact of caring for family members receiving palliative/end-of-life care. Although the review identified
21 studies the evidence was relatively limited.
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Costs were categorised into three main areas: work related costs (costs related to changes in the
caregivers employment), out-of-pocket expenses (direct outlays of money by caregivers) and carer
time costs (costs related to time investment required by carers).The evidence suggested that carers
may give up work, reduced hours at work or used annual leave or sick leave to cope with the
demands of caregiving. Out-of-pocket expenses include costs of medical equipment, prescription and
non-prescription drugs, nursing home expenses, private home care, travel expenses and child care
costs and varied by country. The cost the time investment required by any carer of a patient at the
end of life is significant. The proportion of total care costs met by informal caregivers varied widely in
their reported costs, with estimates of caregiving ranging from 26.6% to 80% of total costs, with wide
variation across different countries. Severe caregiving burden was experienced by many families as a
result of financial issues. Major life changes were often required including moving house, delaying
education or delaying medical care for other family members. Other outcomes included increased
worry, difficulties coping, family conflict and caregiver strain (Gardiner et al, 2014).
Model level of detail – The level of detail included in the model for each resource was limited
primarily by the availability of quantitative evidence, but also by the judgement of stakeholders
engaged in the elicitation process. Thus for example the total cost of emergency hospitalisations per
palliative care patient does not differentiate between the number or type of admissions per patient, it
is therefore not possible to use the model to estimate some suggested impacts of the interventions.
Model analysis – The model predicts the marginal cost impact of the reinforced home care model
compared with a usual home care model. The elicitation exercise was used to obtain judgements
regarding the marginal impact of the reinforced carer support interventions on different categories of
resource usage. The broad range of judgements was then used to obtain a probabilistic description of
uncertainty regarding model input parameters associated with each resource category. Uncertainty is
then analysed using standard probabilistic sensitivity analysis methods. The absence of a single utility
outcome measure means that value of information analyses are not feasible.
Expert elicitation of resource impact of reinforced carer support
A questionnaire was developed examining the cost of the COPE intervention, and the estimated
impacts of the intervention on the components of the palliative care system. A full description of the
elicitation exercise including a summary of the experts’ discussion for each model parameter is
provided in Appendix 27. The elicitation exercise informed the design and parameterisation of the
economic model. Unit costs were taken from routine sources including PSSRU (Curtis, 2013).
Intervention Cost
The cost per patient of the intervention is based on the description in the COPE papers and manuals,
with unit costs.
Visits: 3 visits, total time = 2 hours, Band 6/7 nurses. Cost £130.
Sitting service: Paid sitting support service. Cost £60.
Telephone support: 3 phone calls. Cost £81.
Additional costs: Manuals and training not costed.
Total cost: Estimated cost per patient: £271 per patient
This cost assumes the intervention represents new activity over and above that already provided by the community home care team. Furthermore it may be feasible for trained volunteers to undertake the sitting service reducing this component.
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Impact of the intervention on resource use for the carer
We asked stakeholders their opinion on whether the improved carer burden might translate into
changes in the use of NHS and social service resources by the carer. Stakeholders were consistent in
suggesting that increased carer support might be expected to reduce longer term resource usage for
the carer but all felt that this was hard to identify with any level of reliability.
Impact of the intervention on patient resource use
Expert assessed what type of resource may be impacted by carer support interventions. The
economic evaluation includes all ‘potential’ impacts of the COPE intervention but excludes ‘minimal’
impacts. There was broad agreement amongst participants on the potential domains of impact of the
intervention. Potential impacts were suggested for hospital admissions, Band 6 and 7 Specialist
Nurses, GP consultations, and care home fees. Minimal impacts were suggested for; District Nurse,
GP consultations, Specialist Palliative Care Consultants, Allied Health Professionals incl. occupational
therapist (OT), physiotherapist, dietician, counsellors and psychologists etc., community or hospital
pharmacist and care assistants in health and social care and domiciliary/home care and the cost of
equipment and adaptation to place of residence.
Parameter estimates used in the model based upon literature or elicitation are presented in Table 3.7,
a brief summary of the discussion for each domain focusing on aspects of complexity is presented
here, a fuller description of the discussion including graphs showing individual expert valuations is
included in the Appendix 27.
Table 3.7 Elicitation parameter estimates and unit costs
Parameter Distribution Mean 95% Credibility
Interval
Expert Elicitations
Average duration of reinforcement (%age 90 days) Beta 0.67 0.41 0.88
Proportion avoiding emergency admissions Beta 0.22 -0.08 0.50
Band 6/7 Specialist Nurse: Baseline contact time (hrs/wk.)
Lognormal 1.67 0.66 3.53
Band 6/7 Specialist Nurse: Intervention effectiveness relative contact time
Normal 0.80 0.65 0.95
Proportion of Band 6 Specialist Nurses in team Beta 0.62 0.35 0.85
Care home: Proportion that have average time in care home reduced from 6 months to 3 months
Beta 0.10 0.03 0.22
Care home: Baseline duration of care home stay potentially delayed by 50% (wks.)
Normal 19.50 13.13 25.87
Proportion of GP consultations avoided Normal 0.00 -0.15 0.15
Proportion of GP out of hrs consultations avoided Normal 0.25 0.01 0.49
Proportion of home care population eligible and able to receive reinforced carer support
Beta 0.30 0.09 0.55
Average GP consultations in last 90 days Normal 9.70 6.85 12.55
Average GP out of hrs consultations in last 90 Days
Normal 6.50 5.23 7.77
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Unit Costs: PSSRU and Nuffield EOL Care Costs (£)
Band 6 Specialist Nurses hourly rate Normal 49.00 44.20 53.80
Band 7 Specialist Nurses hourly rate Normal 90.00 81.18 98.82
Home visiting clinical support worker Normal 30.00 27.06 32.94
Telephone consultation Normal 27.00 24.35 29.65
Care home fees Lognormal 605.59 418.72 847.93
GP consultations Normal 45.00 40.59 49.41
GP out of hours / on call Normal 114.00 102.83 125.17
Duration of palliative care in the population eligible for reinforced carer support
The average duration of palliative care following introduction of reinforced carer support depends
upon timely referral of patients for palliative care. There is little evidence regarding typical durations of
care and experts expressed the opinion that this is highly variable, being subject to local service
peculiarities and patient case-mix, specifically in terms of underlying illness and its chronic nature.
Estimates were based upon patterns of referrals to the St Luke’s Community Home Care team.
It was highlighted that duration of palliative care is subject to adaptation; a move towards earlier
referral to care services generally would benefit patients and would allow earlier implementation of the
reinforced carer support intervention, potentially allowing additional benefits to be accrued. It is
therefore possible that this duration may increase as part of the service developments.
Hospitalisations
The analysis focuses on impact of the intervention on emergency hospital admissions. Experts
suggested that a high proportion of emergency admissions arose from acute incidents, commonly at
night or out of working hours, where the A&E department provides the only source of immediate
support for both diagnosis and provision of the care needed. Further, once in hospital many factors
lead to difficulties in gaining discharge and consequently contribute to the high rate of deaths in
hospital.
It was commonly suggested that the key to avoiding recourse to an ambulance call out and
emergency admission was to develop a system that recognises and responds appropriately to acute
crises, confidence between all parties being a crucial factor. It was felt that reinforced carer support
and specifically the COPE intervention had a useful role to play in developing such a system, in
particular “it’s not the Coping skills or the Optimism, but the Planning and Expert information” that will
have the greatest impact.
Band 6 & 7 Specialist Nurses
Four of the seven experts felt able to provide information on the baseline specialist nurse input into
care, primarily based upon the working practices and patient population experienced by the St Luke’s
Sheffield team. All participants agreed that the reinforced carer support intervention would be likely to
have an impact on the patient/carer contact time, though this impact might vary according to the
intensity of care needs and would be dependent on the skill mix/grade of nursing carers employed.
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GP costs
The average number of GP consultations is derived from studies by the Nuffield Trust (Georghiou,
2014) and The Choice in End of Life Care Programme Board (2015) costing study. The experts were
consistent in the opinion that the prime effect GPs would be through avoiding out-of-hours or on-call
consultations.
The experts identified a complex set of interrelated factors that may both moderate and accentuate
effects of the intervention on GP resources. Inadequate availability or signposting of supporting
services has the potential to nullify intervention effects. The intervention may impact on aspects of
care such as psychological support more than pain control, though concurrent deliver may limit
effects. Reinforced carer support may alter the quality and content of GP in and out-of-hours
consultations rather than the number of or duration of consultations.
Care home fees
Most experts judged that reinforced carer support may delay or prevent transfer of patients into a care
home setting, though lack of clarity regarding pathways for patients entering care homes during the
last few months of life means that estimates are highly uncertain with further research warranted.
Population receiving the reinforced carer support intervention
Only a certain proportion of the palliative home care patients and carers will be able, eligible or willing
to take part in the reinforced carer support intervention. Stakeholders noted that changing
demographics mean that the availability of carers is reducing. The identification and recruiting of
carers is also dependent on indeterminate service characteristics such as the ability to predict the
remaining life course of patients and the development of confidence between patients, carers and
care professionals.
Varied targeting strategies were discussed by the experts, with most suggesting targeting patients
who would benefit most. Equity considerations imply offering additional carer support to all carers, or
identifying alternatives that may be available for those not willing or able to take up the intervention.
The ability to estimate the total potential workload was limited by variation in the definition of the home
care population, which may have a very different case mix pattern from one geographical service to
another.
Model Implementation
The expert elicitation exercise informed the design of the economic model. A simple tabular
spreadsheet design was used to estimate impacts on a range of economically relevant outcomes. The
absence of a well-defined single index utility outcome measure precluded the use of a standard
decision analytic approach.
Whilst a probabilistic approach is used to characterise parametric uncertainty in the model, the
absence of a single utility outcome prevented a value of information analysis based on expected
opportunity loss associated with decision. The results of the elicitation exercise were used within the
economic model to predict the potential cost impact of the introduction of reinforced carer support.
Results of economic modelling
The key health and wellbeing outcomes for carers and patients, as identified by the review of
effectiveness (Burns et al 2015) are summarised below. The economic model considered the
marginal resource and cost impacts only of introducing reinforced carer support into an existing home
based model, Results are presented in terms of cost per patient, resource implications and overall
budget impact on the whole and constituent parts of the palliative care system.
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The results of the economic modelling work need to be considered in conjunction with the health and
wellbeing outcomes summarised below.
Patient and carer health and wellbeing outcomes
Effectiveness for patients: Across patient outcomes most of the measures were consistent, showing
that reinforced home-based palliative care interventions were neither significantly better nor worse
than non-reinforced home-based care with respect to patient outcomes. Two studies measuring
symptom control showed positive effects and three studies showed neutral effects. Psychological
health outcome were positive in two studies and neutral in two studies. Other outcomes were neutral
in all studies which measured them: patient QoL in three studies, patient pain in one study,
hospitalization in two studies, patient response in two studies and patient satisfaction with care in one
study.
Effectiveness for informal caregivers: Across caregiver outcomes, the majority of interventions
showed no effect. For caregiver QoL, nine measurements across eight studies showed four positive
and five neutral effects. Sixteen estimates for psychological health (eight studies), of which three
favoured the intervention, one favoured the control, and twelve were neutral. Response outcomes for
lay caregivers were measured fourteen times (six studies), yielding four positive and ten neutral
effects. Caregiver satisfaction with care, measured only once, showed a neutral effect.
Health and social care outcomes - Cost per patient
The estimated changes in operating costs consequent on introducing reinforced carer support, based
upon the expert elicitation exercise are summarised in Table 3.8.
The reinforced carer support intervention was estimated to cost between £250 and £300 per patient.
However it was projected that impact on Band6/7 specialist nurse costs meant that the marginal cost
to the palliative care service would be in the region of £90 per patient (-£230, £240). Throughout the
health and social care system however the intervention was potentially cost saving with an average
marginal cost saving of £560, varying between a saving of £1440 and an increased cost of £170. By
far the largest component of projected cost savings arose from avoiding inpatient stays resulting from
emergency admissions to hospital.
Table 3.8 Summary of changes in costs associated with reinforced carer support
Palliative care service Hospitalisation costs (Marginal)
GP costs (Marginal)
Care home costs (Marginal)
Total marginal cost
Cost of intervention
Band 6/7 Specialist Nurse
Change in PC costs
Mean £270 -£180 £90 -£510 -£120 -£10 -£560
Lower 95% CI
£250 -£510 -£230 -£1,340 -£280 -£30 -£1,440
Upper 95% CI
£300 -£30 £240 £160 £0 £0 £170
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Health and social care outcomes - Resource implications
Table 3.9 presents the implications for health and social care resource requirements. The model
predicted that the overall number of hours of band 6/7 specialist nurses may be reduced, if the
predicted reduction in nurse visits following successful implementation is achieved.
Health and social care outcomes - Budget impact
Table 3.10 presents the total budget impact across the whole palliative care system of implementing
the intervention in a range of population sizes.
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Table 3.9 Health and social care resource implications
New palliative home care patients per month
Carers receiving the intervention
Band 6/7 Intervention workload (hrs/month)
Change in Band 6/7 operational workload (hrs/month)
Change in Band 6/7 hours
Marginal Band 6/7 FTE
Marginal GP consultations
Marginal care home weeks
25 7 (2,14) 23 (7,42) -33 (-102,-4) -10 (-70,19) -0.06 (-0.43,0.12) -1.1 (-2.7,0.4) 0.02 (0.01,0.06)
50 14 (4,28) 46 (14,84) -66 (-204,-8) -20 (-140,38) -0.12 (-0.86,0.24) -2.2 (-5.4,0.8) 0.04 (0.02,0.12)
100 28 (8,56) 92 (28,168) -132 (-408,-16) -40 (-280,76) -0.24 (-1.72,0.48) -4.4 (-10.8,1.6) 0.08 (0.04,0.24)
200 56 (16,112) 184 (56,336) -264 (-816,-32) -80 (-560,152) -0.48 (-3.44,0.96) -8.8 (-21.6,3.2) 0.16 (0.08,0.48)
Table 3.10 Budget Impact
New palliative home care patients per month
Monthly cost of providing the intervention (95% CI)
Change in palliative care service cost (i.e. net of Band 6/7 nursing) (95% CI)
Change in hospital costs (95% CI)
Change in GP costs (95% CI)
Total monthly marginal cost (95% CI)
25 £2,000 ( 600, 3900) £610 ( -1830, 2380) -£3,760 ( -11710, 1200) -£940 ( -2640, 0) -£4,190 ( -13260, 1180)
50 £4,000 ( 1200, 7800) £1,220 ( -3660, 4760) -£7,520 ( -23420, 2400) -£1,880 ( -5280, 0) -£8,380 ( -26520, 2360)
100 £8,000 ( 2400, 15600) £2,440 ( -7320, 9520) -£15,040 ( -46840, 4800) -£3,760 ( -10560, 0) -£16,760 ( -53040, 4720)
200 £16,000 ( 4800, 31200) £4,880 ( -14640, 19040) -£30,080 ( -93680, 9600) -£7,520 ( -21120, 0) -£33,520 ( -106080, 9440)
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Discussion
The COPE reinforced carer support intervention is estimated to cost between £250 and £300 per
patient. Experts’ judgments suggest that the intervention may be associated with cost saving of £560
(ranging between saving £1500 and an increased cost of £170) per patient, with the largest
component of projected savings arising from avoiding emergency admissions to hospital. The
economic analysis is heavily reliant on expert judgment and represents an exploratory analysis, fit for
the purpose of informing the design of further research and/or the design of an adaptive palliative care
service. The use of this analysis to inform decisions to purchase (or not) reinforced carer support
would be subject to many provisos.
The economic analysis focused on the COPE intervention, a theoretically-grounded brief intervention
teaching problem solving skills to carers to assess/ manage patient symptoms. Other reinforced carer
support interventions may have different aims and strategies (e.g. offering self-care techniques and
psychological support or offering needs assessment or helping to mobilise support networks). Where
interventions operate via alternative causal chains the generalisability of the economic results may be
limited.
Reinforcement interventions of the COPE type, furthermore, may vary according to timing of the
intervention (early versus last weeks of life), duration of the intervention (limited or on-going support),
the type of support offered (e.g. assessment of need and goal planning or generic education/training).
Stakeholders highlighted that these factors all had the potential to impact on effectiveness and costs.
Our analysis assumed introduction of the intervention at around two months prior to death of the
patient but exploration of the benefits of earlier implementation is needed.
Stakeholders stressed that the effectiveness of the intervention, particularly on avoiding hospital
admissions, was highly dependent on the state of the wider health and social care system. For
instance, imperfect communication between key palliative care agents, lack of access to out of hours
services and limited access to patient records would all limit the ability of the carer support
intervention to prevent avoidable admissions. During the validation process it became apparent that
the expert judgements concerning potential impact of the intervention tended to assume an optimally
operating health and social care system. This 1) implies problems in generalising from one setting to
another and 2) suggests a central difficulty in defining the intervention as being either the specific
reinforcement component (for example the COPE carer educational support component) or the entire
home palliative care system including the reinforcement component.
The quality adjusted life year (QALY), the single health utility outcome preferred by health economists
to support resource allocation decision making is rarely used in the palliative care field. Particularly
the domains captured in the generic QALY instruments such as the EQ5D and SF36 are insufficient
to capture important outcomes of palliative care. Many inconsistent outcomes and outcome measures
are currently in use in palliative care. For reinforced carer support the outcomes of both the carer
themselves and the patient they are supporting are important. For carers of cancer patients the COPE
intervention improved burden related to patient’s symptoms and caregiver task burden, although
many of the other outcomes measured showed no significant change. Further research is needed to
explore the outcomes that are most appropriate for the multiple perspectives, and how to balance
these, especially if they are conflicting.
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Reflections
We reflected on issues of complexity, integration, stakeholder involvement and the value and
application of the methods developed for effectiveness HTA.
Complexity
The palliative care system and specifically reinforcement interventions in palliative care demonstrate
all the described aspects of complexity, including multiple perspectives from the many different
stakeholders, indeterminate phenomena, uncertain causality, unpredictable outcomes and time/path
dependence (historicity).
Problems with the generalisability of the economic case study were highlighted at many different
levels of the analysis, including interpretation of effectiveness and resource use evidence, obtaining
relevant, meaningful and specific descriptions of current palliative care practice and the reinforcement
intervention and in the interpretation of the economic results of the evaluation. These problems in
generalisability affect the relevance of the analysis to international, national and different local
palliative care settings and indeed between one implementation of the reinforcement intervention and
the next.
To a certain extent these problems of generalizability can be alleviated by improved clarity in the
reporting of the conditions of an evaluation or trial, for instance by developing and abiding by more
rigorous reporting standards that require the details of an intervention to be reported (Pinnock 2015).
It might be expected that improving reporting of studies will improve our understanding of the diversity
and variation within system settings and interventions and allow us to make informed judgements
about the relevance of evidence and potentially even support quantified adjustments to estimated
impacts between settings. However, where this lack of generalisibility arises from indeterminacy
within the system, from uncertain causality and from inherent historicity, there will be a limit to what
can be achieved from improved reporting. In these circumstances there will inevitably be a gap
between evidence generated in one setting and its applicability to implementation in another. In
essence these problems are an unavoidable consequence of the complexity of the system and
intervention. Indeed these problems with generalizability may be considered as defining aspects of
the complexity of the system and intervention, such that if there are no problems of generalisability it
is questionable whether the intervention is truly complex as oppose to complicated.
One possible response to the gap between evidence and implementation is to require that the novel
complex intervention includes an integral mechanism for continuous evaluation and intervention
adaptation. Thus for example in considering reinforced palliative care, this study described the
intervention as comprising the introduction of a specialist palliative care nurse to provide carer
training. In addition the intervention should include the mechanisms for monitoring and evaluating
outcomes achieved and for redesign of the reinforcement intervention to enable health and wellbeing
and economic outcomes are achieved. These mechanisms would necessarily include the
specification of a data set for enabling that evaluation of outcomes.
Integration
The conceptual modelling phase of the economic evaluation used the logic model for palliative care
as its starting point. The problem oriented conceptual modelling for the economic model expands on
the thinking within the logic model by developing descriptions of the health systems that enable the
potential impact of the intervention on economically relevant outcomes to be made explicit.
77
There is overlap between the conceptual modeling work for the economic model and other elements
of the project. During the development of the economic model we worked closely with those
undertaking the effectiveness review. Ideally closer ongoing interaction with other elements, including
the socio-cultural and context and implementation work programmes, would have allowed us to gain a
more rapid and comprehensive understanding of these issues to feed directly into our conceptual
modelling exercise.
Stakeholder input
The input of stakeholders at all stages of the modelling process, from scoping to interpretation and
validation of results, was vital in gaining a description of the potential causal relationships within the
system. Several modes of stakeholder engagement were involved in the economic case study
including email communication, telephone interviews, face to face meetings, and workshops.
Engaging with stakeholders is a resource intensive exercise both for the stakeholders and the case
study team. The enthusiasm and commitment to engage and stay engaged is highly dependent on
the relevance of the decision problem. (See Appendix 28 for a summary of stakeholder feedback
about their involvement in the economic analysis).
The high level of indeterminacy in the palliative care system meant that interactive forms of
communication were the most productive forms of stakeholder engagement. Telephone and face to
face meetings allowed relevant background material to be provided flexibly and responsively to
participants to ensure a common level of understanding. Secondly, these approaches meant that we
could collect sufficient discussion to enable an appropriate interpretation of the participants’
responses. Thirdly we could collect information flexibly, that is eliciting information from each
participant in a form that they were able to make a judgement about, yet sufficiently coherently across
participants to enable meaningful synthesis. The workshop environment further allowed a shared
understanding to be developed amongst stakeholders. This shared understanding being an important
basis to ensure that the quantitative model reflects the different understandings of the system and yet
allows the modellers to construct a coherent and credible synthesis that is fit for decision making
purposes.
In this case the reliance on subjective judgement arises from the gap between the formal evidence
base and the needs of decision makers; this is potentially a generic feature of complex systems
arising from characteristics such as the dynamic nature of such systems or the inability to adequately
describe complex social systems from any single point within. This is not to say that research in these
systems is futile but to recognise that decision making needs are likely to continue to outstrip the
evidence base in many complex phenomena. This use of the conceptual modelling approach to
uncover stakeholders understanding, generate debate and develop a description of the system that
reflects both agreement and differences reflects a hermeneutic position that recognises that meaning
is created through intersubjective communication. In this case it is this interaction between the
modeller and stakeholders in developing mutual understanding of a model that establishes a model’s
significance and its warranty for decision making.
Value and application of methods
The economic component of the case study tested the use of a systems approach, using a
combination of problem structuring methods, reviews of existing evidence and quantitative modelling,
for undertaking model based economic evaluation of a complex intervention in a complex
setting.(Chilcott et al 2015).
The lack of evidence on the cost and resource impacts of the reinforcement interventions was
compounded by a high level of indeterminacy in the structure of the system, for instance, the absence
of agreed taxonomies, definitions, outcomes and outcome measures for many parts of the system.
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The case study has demonstrated the ability of the guidance to facilitate the development of an
exploratory economic model in complex situations where the evidence base is at an early phase.
In particular the first two steps of the systems approach – understanding the decision problem and the
conceptual modelling phase – have contributed to a sophisticated understanding of the palliative
home care system and raised important issues concerning the economics of interventions within this
setting and the design of evaluative research.
Existing reviews of the cost effectiveness of interventions within palliative care have tended to group
palliative interventions together either as one large group, or sometimes, by types of primary care
setting (e.g. home care, hospital care) in an attempt to answer the question of whether or not
palliative care is cost effective. This approach fails to take into account the complexity of the
interventions and the palliative care system. By following the guidance we sought to understand the
decision problem in a way that captured the views and perspectives of the different stakeholders. We
then developed an explicit description of the health system recognisable to stakeholders and used
this as the basis for subjective estimates of the potential impact of the intervention(s) on economically
relevant outcomes. This allowed the analysts and stakeholders a better understanding of the
differences between superficially similar interventions and the issues which arise when attempting to
implement these within the palliative care system. It also gave an appreciation of the potential
differences in the baseline services and how these might impact on the effectiveness and economic
impact of the intervention. Although the economic model itself was ultimately limited by the lack of
evidence within the field, the conceptual modelling provides a robust basis from which to design future
research.
Conclusions regarding economics
Palliative care is a complex intervention, which provides many of the conceptual and methodological
challenges typically associated with the assessment of complex health technologies. To date the
application of health economics in the field of palliative care has been limited. No existing economic
evaluations of reinforced home palliative care have been identified and only a limited number of
evaluations of (non-reinforced) home care exist.
We have undertaken an exploratory analysis of the economic impact of introducing a carer support
intervention into an existing home care service, based on the elicitation of expert judgments. The
model development followed the five phase process outlined by the guidance for using a systems
approach for economic modelling of complex interventions in complex settings. The COPE reinforced
carer support intervention is estimated to cost between £250 and £300 per patient. The intervention
has the potential to impact on the use of other health and social care service, importantly emergency
hospitalisations. The expert judgements suggest that this may be expected to result in cost savings
with a predicted average marginal cost saving of £560 (-£1500, £170) per patient receiving the
intervention.
The generalisability of this analysis to other settings will necessarily require careful interpretation. For
instance characteristics specific to the UK system, for instance the funding system and historical
initiatives such as the introduction of the 2003/4 ‘GP Contract’ which removed GPs’ 24hr responsibility
for the patient will have an influence on the effectiveness of any new intervention within it. Locally the
level of co-ordination of and communication between different services has the potential to undermine
or enable intervention effects.
The results of this economic modeling exercise are exploratory and highly uncertain. This exercise
does however provide a detailed understanding of the palliative care system and how a reinforced
79
carer support intervention may impact on this system. The implementation of the model was hindered
by the lack of evidence.
The conceptual modeling and the exploratory results based on this elicitation exercise provide are
aimed at providing a useful basis for the design of future research and to provide the economic
evidence to support decision making in this field.
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Ethical issues in home and reinforced home based palliative care
Aim
To assess ethical issues of reinforced and non-reinforced home based palliative care services
(HBPC) using guidance developed in the INTEGRATE-HTA project1.
Methods
We focused on identifying strengths and weaknesses of existing methods for the assessment of
ethical aspects for complex health interventions. The resulting information is included in a procedural
framework, which forms the core content of the guidance on ethical issues (Lysdahl, Mozygemba,
Burns, Chilcott, Brönneke, Hofmann, (Eds.) 2015). The framework consists of a step based ethical
analyses process, and two contextual areas (the HTA/ethical approach context and technology
context) that influence, and are influenced by, the steps in the analyses (see Figure 3.5). In addition to
guiding the choice of an ethical approach, the framework guides amendments needed in the
application of the ethical approach.
The framework was applied in the case study by one researcher with knowledge and insight into
palliative care (not an HTA expert and not a trained ethicist)2. Literature describing the specific
selected approach (recommended in the guidance) was used in the application. The researcher used
‘reflective thoughts’ to draw on existing knowledge3, known literature (e.g. Haughey C & Watson M,
2015), and the advice provided by stakeholders, i.e. patients, relatives, professionals, academics and
other involved parties during stakeholder consultations at the start of the project.
Stakeholder meetings focusing separately on ethical issues were not arranged because the
framework for ethical analyses regards stakeholder involvement as a "common exercise" in the HTA
process (i.e. the information provided by stakeholders will not be limited to only one aspect of the
HTA, but cover more/all aspects).4 Hence, it was considered acceptable and advisable to let the
outcome of stakeholder meetings arranged for other aspects of the project e.g. the economic
analyses to inform the application of the ethical guide in the case study. The initial stakeholder
meetings in this project used this common approach, and the ethically relevant objectives and issues
identified from this exercise were also used in the application (see step 2 of this case study).
Throughout the application process, consultations between the researcher applying the guidance and
the developers of the guidance were occasionally also needed. After applying step 3 and the first part
of step 4 the (first) author of the guidance assisted in summarising the moral relevant questions.
1 A set of 7 specific questions were addressed in the application process for structuring the feed-back, further details are
provided in 'Guidance for assessing effectiveness, economic aspects, ethical aspects, socio-cultural aspects and legal aspects in complex technologies' (see Lysdahl KB, Brereton L, Oortwijn W et al (2015) Guidance to assess ethical aspects In: Lysdahl, KB, Mozygemba, K, Burns J, Chilcott, JB, Brönneke, JB, Hofmann (Eds) (2015) 'Guidance for assessing effectiveness, economic aspects, ethical aspects, socio-cultural aspects and legal aspects in complex technologies. [Online]. Available from: http://www.integrate-hta.eu/downloads/
2 This was in accordance with the overall idea that the application of simulating “an ordinary HTA setting” as far as possible.
3 Reflective thoughts involved the researcher actively and critically thinking to draw on knowledge and experience of
palliative care as well as information provided by stakeholders when applying the framework. The intention was to develop
an incremental understanding of the complexity of palliative care and ethical issues and communicate this to other
researchers.
4 The resource considerations is an obvious reason for this. Besides, we may assume that relevant ethical issues will be
raised by stakeholders, even if the focus and purpose of the stakeholder involvement differs.
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As Figure 3.5 shows, we recognised close links between ethical, legal and socio-cultural issues in
HTA. Discussions between researchers during the assessment of ethical and socio-cultural issues
guidance, revealed common topics arose relating to HBPC. This indicates possible benefits of close
collaboration when dealing with these aspects, which turned out to be beyond the scope of the
demonstration HTA to fully explore.
Figure 3.5 Framework for ethical analysis in HTA of complex interventions
Findings
From step 1 important aspects of complexity5 for HBPC were identified (see Table 3.5 and appendix
24) and then presented in order of perceived importance to the researcher applying the guidance as
ordering the importance facilitates the next step of the application.
1. The indeterminate nature of HBPC and conditions appears to be a key issue as the provision
of informal carer support needs to be individually tailored to meet the informal carer and
patient’s needs. Indeed, the holistic philosophy of palliative care and the interrelatedness of
patient and carer needs means that both home and reinforced models of home palliative care
will vary with regard to the dyad’s needs, their roles and requirements for access to available
services. Service access may vary geographically. Additionally, there is a lack of clarity about
where the reinforced intervention ‘sits’ within service provision and whose responsibility it is to
implement carer support in a timely manner (especially as carers may have been providing
care for patients with life limiting illnesses as part of rehabilitation services.
5 The analysis is based on the complexity characteristics presented in a submitted manuscript by Lysdahl and Hofmann: ’Complex
interventions: Characteristics relevant for ethical analysis in health technology assessment’, characteristics which are also included in the guidance by Lysdahl KB, Mozygemba, K, Burns J et al (Eds.) (2015).
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2. The multiple, changing perspectives of carers and patients are important considerations,
especially as caregiver support may be intended to change perspectives (e.g. with regard to
perceptions of burden or their own caregiving competence).
3. Widespread ethical complexity exists in relation to both home and reinforced models of home
palliative care provision.
4. There is a lack of consensus about the outcomes that should be assessed and the
instruments used to measure these.
5. Uncertain causality is evident given the lack of consensus about the outcomes that should be
assessed and the instruments used to measure these.
By applying the different available tools in step 2 (Lysdahl, et al 2015), the ethical approaches were
assessed for their suitability for HBPC in the context, which resulted in a final selected ethical
approach to use in the subsequent application 6. These are
Interactive, participatory HTA approaches appeared to ‘best fit’ the complexity profile of
reinforced HBPC, which was considered complex in terms of all five characteristics of
complexity. However, Social Shaping of Technology and Interactive, participatory HTA
approaches are very similar in terms of their ability to deal with aspects of complexity.
Assessment of how the ethical approaches fit with the integration perspective of the HTA
commissioner/agency favoured the four following ethical approaches: principalism, casuistry,
the triangular model and axiological approaches (Socratic, EUnetHTA). These are all
applicable when the ethical analysis is a subsumed part 7 of the HTA, which is in accordance
with the integration perspective of the National Institute for Health and Clinical Excellence
(NICE) commissions health technology assessment in UK.
Assessment of how the ethical approaches fit the local HTA context policy of aiming for
assessment versus appraisal indicated that the Axiological approach was most applicable.
This approach is most applicable when the aim of the HTA is assessment not appraisal, like in
UK where the Health Technology Assessment (consisting of a systematic review of the
evidence and an economic evaluation) is separated from the work of the appraisal committee.
Balancing these assessments against each other led to the selection of the Socratic approach
as the most suitable for assessing ethical aspects of HBPC in the local context.
The ethical approach selected in step 2 may not fit important complexity aspect of the technology as
other considerations are taken into account. In step 3 possible shortcomings of the selected approach
is identified, so desirable or necessary amendments or additions can be made. The assessment tool
in the guidance points to possible shortcomings of the Socratic approach, which is relevant for HBPC
the following way:
Addressing the moral impact of indeterminacy, which in case of palliative care relates to the
the diverse range of home care models that exist (in UK), and the lack of understanding of the
definition & nature of palliative care by professionals, patients and the public/society.
6 See guidance for more details about the process on how to apply these tools in the ethics section of the guidance by Lysdahl KB,
Mozygemba, K, Burns J et al (Eds.) 2015. 7 When ethics is a subsumed part of the more comprehensive HTA it is regarded “a subsidiary activity, a sub-project of an HTA,
resulting in a separate (subordinate) chapter in a HTA publication/report. Both the activity and the end result (chapter in report or published article) may be less important in the subsequent decision making process.” (Hofmann et al. 2015)
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Subsequent moral impact of this concerns access independent of type of disease, and
avoidance of futile treatment.
Dealing with the multiple perspectives of the many professional and lay stakeholders involved
in HBPC, including the ability to deal with issues of decision making and responsibilities, both
of which are essential elements in HBPC where decisions need to be made throughout the
illness trajectory by various stakeholders with different roles and responsibilities.
Although HBPC is associated with many outcomes and a lack of standardised outcome
measures, the Socratic approach is limited in its ability to address a variety of outcomes.
Uncertainly of outcomes may relate to adequacy of professional and carer training, a concern
that may not be covered by the Socratic approach.
In step 4 the Socratic approach was applied to HBPC, which included to assess the relevance of 33
(listed) moral questions for HBPC and justify the selection.8 The outcome of this, together with the
identified shortcoming of the approach (described above) was then summarised into the 6 broader
ethical issues relevant for rHBPC. In the description of these below, the important morally related
aspects (within these 6) are highlighted in bold letters. The 6 issues are also largely interlinked, some
indications of this is given in square brackets.
Vulnerability
Patients requiring HBPC are vulnerable for many reasons: stakeholders reported that palliative care is
associated with stigma, reduced independence, lacking ability of practitioners to recognise patients’
palliative care needs, under diagnosis of palliative care need may result in under treatment of pain
and symptoms and over treatment in terms of exposing the patient to futile treatments. It is important
to improve understanding of the definition and nature of palliative care by professionals, patients and
the public / society, especially as a diverse range of home care models exist.
Changing roles and relationships
Home based care changes the patient’s roles within their existing relationships as the individual
becomes the recipient of care from an informal caregiver (who may be a spouse or family member).
The individual’s change of role to care-recipient is likely to be accompanied by change in status (both
within and outside of the family unit as a result of changes in ability, financial income and perceived
social status).
Similarly informal carers will experience a change of role, financial income, loss of employment
(they often cannot manage employment alongside their caregiving responsibilities) and status as a
result of undertaking home care. Carers too may suffer from stigma associated with change in role
and status. The value of adopting a caregiver role may change for the informal caregiver if they
experience caregiving as burdensome and derive few satisfactions from the role. There is a risk of
potentially becoming patients themselves as a result of the burden experienced through caregiving
[i.e. contributing to iatrogenic illness].
8 This approach do itself consist of steps, of which the two first (Identify the intended purpose of the health technology and reveal the
background for the assessment (1) and Identify the involved persons, groups, and stakeholder (2)) was considered covered in the initial steps of the application. As already indicated the final steps of the Socratic approach were not applied, hence the outcomes are preliminary. This preliminary outcome was also fed into the executive summary template for WP6 integration exercise.
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The presence of an informal carer in itself changes the relationship between patients and health
care professionals. Although the philosophy of palliative care is to view patients and their family as
the ‘unit’ of care, the adoption of a caregiver role by family members also changes the family
member’s relationship with health professionals who may view them as a co-worker. Concerns are
raised about the adequacy of professional and carer training (and therefore competence to provide
palliative care).
Unclear responsibility is a key issue, regarding who has ultimate responsibility for patient care
(particularly at night) in the current system, and who has responsibility for patient and carer needs
assessment.
For professionals: key issues that require careful consideration as a result of care being provided
within the patient’s home relate to ensuring privacy and maintaining confidentiality as care records
will be kept within the patient’s home. [Cf. vulnerability and shared decision making]
Diverse (interacting) perspectives and shared decision making
The intention of HBPC is to support both patients and informal carers, thus enabling patients to stay
and, if preferred, die at home by providing physical and psychosocial care that maintains their quality
of life.
There may be differences in patient and informal carer choice/preferences (e.g. the patient may
prefer to be cared for at home, but the informal carer may prefer care to be provided in an institution).
This can result in a conflict of interest between patients and carers.
Hence, ensuring patient and caregiver autonomy, self-determination and valid consent for both
patients and carers is challenging given their different needs. Effective communication to allow
patients and carers to retain their participation in decision making and a sense of control over their
share of decision making is important.
Patients and informal carers need to be fully aware of the patient’s prognosis (not only death but the
process of dying). Raising awareness of dying requires professionals to be sensitive to the verbal and
non-verbal cues of patients and informal carers indicating their readiness to deal with the subject.
Professionals will need to respect individual preferences, cultures and values. Sensitive,
compassionate, honest, open, understandable communication between patients and families and
staff is a crucial element of care and one that underpins informed consent. [Cf. changing roles and
relationship and voluntariness]
Voluntariness
Key issues of concern around informal caregiving lie in ensuring that caregivers are willing, (not
coerced) to undertake the caregiving role and make an informed decision about adopting the role and
have the ability to care at home. Informal caregivers may feel morally obliged to take on and / or
continue in a caregiving role as a result of promises made to the patient (e.g. to fulfil wedding vows or
promises made during the illness period).
The implementation, use, and withdrawal of HBPC challenges patient and carer autonomy as the
result of communication difficulties resulting from a variety of causes (e.g. patient capacity, physician /
staff paternalism or restrictions on choices available). [Cf. vulnerability and shared decision
making]
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Access and availability
There are known issues regarding access to palliative care and HBPC, resulting from concerns about
the lack of availability of services and specific professionals. This raises questions about fairness /
equity of access / just distribution of resources. Important to ensure easy (e.g. out of hours), equitable
(e.g. to all groups) and timely (e.g. not restricted only to the last months of life) access to palliative
care, and to provide support for family carers post bereavement. [Cf. vulnerability, under- and over-
treatment].
Outcome uncertainties
Key ethical issues lie in providing adequate pain and symptom (physical, psychological and
emotional) control without creating harm. However, the benefits and harms of the implementation, use
or withdrawal of HBPC are not fully known, especially from a safety perspective for patients (e.g.
medication use off license - outside of the indications for which they are licensed by national
regulatory bodies) and carers. That said, there are known potential physical and psychological health,
financial and social consequences of adopting a caregiver role for individuals who take this on.
This lack of knowledge means that it is not possible to say how the harms be balanced against the
benefits [i.e. the principle of beneficence].
A diverse range of outcomes are measured using a variety of tools meaning that there is little
consensus re choice of outcomes and a range of agendas for measuring outcomes. E.g. the interests
of health providers relate to cost and clinical effectiveness, as well as safety.
[Cf. conflict of interests: which perceptive reflected in the choice of outcome measures]
Summary of findings
The 6 broader issues identified in the application, do not match perfectly with one single category in
the HTA research question of our project, they rather relates to many:
Vulnerability and Changing roles and relationships relates to acceptability,
meaningfulness, and appropriateness.
The same categories are also relevant for Shared decision making and Voluntariness, but
these may also relate to feasibility.
Access and availability acceptability and appropriateness.
Outcome uncertainties match appropriateness and effectiveness, but also relates to cost
effectiveness.
Reflections on the application of the guidance to assess ethical issues
We reflected on issues of complexity, integration, stakeholder involvement and the value and
application of the methods developed for effectiveness HTA.
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Complexity
In contrast to effectiveness, the guidance of ethical aspect is designed exactly to elucidate the
complexity of the intervention at hand, and guide the accompanying questions on how to proceed with
the ethical assessment. Indeed, the application in HBPC revealed a range of features of the
intervention that illustrate its complexity, as summarised in the findings of step 1 of the ethical
assessment (see appendix 24).
Integration
A number of common topics, at least relevant for ethical and socio-cultural aspect, can be found in
HBPC, like ‘Shared decision making and patient-professional relationship’, ‘Changing roles’, ‘Access
and availability’. This is not surprising as these aspects of HTA are interrelated, and often addressed
in common (also with legal aspects, and together labelled ESLI research) in research articles and
guidelines (Lehoux & Williams-Jones, 2007). (Potter et al., 2008) (Braunack-Mayer & Palmer, 2008)
(SBU, 2014). A main challenge highlighted in the literature is to integrate these aspects with
effectiveness and economic aspects. Hence, the ethical guidance recommends collaboration across
assessment aspects for SAP involvement, to share evidence emerging from each application.
Stakeholder input
The involvement of stakeholders in many parts of the HTA process is essential in complex
interventions, as highlighted in the guidance on ethical aspects. As the approach of the application
was to simulate “an ordinary HTA setting” as far as possible, stakeholder involvement focusing
exclusively on ethical aspects was avoided. It is likely that SAP meeting specifically addressing ethical
issues, led by a trained ethicist, may have identified additional ethical issues of HBPC and explored
the values underlying our findings in more depth. However, we consider involving SAPs specifically
for each of the aspect of HTA to be beyond practice in an ordinary HTA practice for resource reasons.
Furthermore, ethical issues will emerge and be included in SAP meetings discussing economical or
socio-cultural aspects of reinforced home based palliative care. For instance the topic access to
services is relevant from an economic perspective (the costs of providing access), a socio-cultural
perspective (group differences in access) and an ethical perspective (distributive justice). This is in
accordance with the understanding of complex intervention where different perspectives and
components are interconnected and interacting.
Conclusion
The application of the guidance on ethical aspects in HBPC did not aim at full ordinary HTA, but
rather a demonstration HTA of HBPC. Accordingly, there are limitations of the application. These
mainly concerns the lack of opportunity to accomplish a full ethical analyses (i.e. carrying out all steps
of the framework) and the artificial, not HTA agency, setting of the application (e.g. lack opportunity to
gain support from an “unprejudiced” ethicist). Hence, the evidence does not allow us to draw any firm
conclusions about morally relevant differences between reinforced and non-reinforced home-based
palliative. Reinforced care may, for instance, enhance shared decisions making. However, in view of
the lack of evidence we cannot exclude that reinforced, on the contrary, hinders shared decision
making, due to changing roles and unclear responsibilities. Nevertheless, the application add value by
highlighting complexity aspects of HBPC, relevant for the ethical analysis, by identifying a range of
ethical issues involved, and by adding arguments for an integrative approach to HTA.
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References
BRAUNACK-MAYER, AJ., & PALMER, N. (2008) Health Technology Assessment: Ethical, Legal and Social Issues. In K. Hegenhougen & S. R. Quah (Eds.), International Encyclopedia of Public Health (3 ed., pp. 265-269): Elsevier.
HAUGHEY, C., & WATSON, M. (2015) Ethics in palliative care. InnovAiT: Education and inspiration for general practice, 1755738015574750.
HOFMANN, B., OORTWIJN, W., BAKKE LYSDAHL, K., et al. (2015) Integrating ethics in health technology assessment: many ways to Rome. International Journal of Technology Assessment in Health Care,:1-7.
LEHOUX, P., & WILLIAMS-JONES, B. (2007) Mapping the integration of social and ethical issues in health technology assessment. International Journal of Technology Assessment in Health Care, 23(1), 9-16. doi: 10.1017/s0266462307051513
LYSDAHL, K.B., BRERETON, L., OORTWIJN, W., MOZYGEMBA, K., REFOLO, P., SACCHINI, D., BRÖNNEKE, J.B., VAN DER WILT, G.J., GERHARDUS, A., HOFMANN, B. (2016b) Guidance to assess ethical aspects. In: LYSDAHL, K.B., MOZYGEMBA, K., BURNS, J., CHILCOTT, J.B., BRÖNNEKE, J.B., HOFMANN, B. (eds.). Guidance for assessing effectiveness, economic aspects, ethical aspects, socio-cultural aspects and legal aspects in complex technologies [Online]. Available from: http://www.integrate-hta.eu/downloads/
POTTER, BK., AVARD, D., GRAHAM, ID., et al. (2008) Guidance for considering ethical, legal, and social issues in health technology assessment: application to genetic screening. International Journal of Technology Assessment in Health Care, 24(4): 412-422. doi: 10.1017/s0266462308080549.
SBU (2014) Utvärdering av metoder i hälso- og sjukvården, en handbok. Etiska och sociala aspekter. Stockholm: Statens beredning för medicinsk utvärdering.
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Socio-cultural aspects9 in (reinforced) home based palliative care ((r)HBPC)
Background
Although the addressing of social aspects is part of most definitions of HTA, this is rarely done in
practice (Arellano et al., 2009; Draborg et al., 2005; Lehoux et al., 2004). Where they are addressed,
this is often done in an unstructured and unsystematic way (Lee et al., 2009). This can mainly be
traced back to two major reasons: the lack of clarity of the concept of socio-cultural aspects as well as
the lack of well-developed methods for the assessment thereof. In the INTEGRATE-HTA project we
worked on both issues.
Aim
We aimed to identify and assess socio-cultural aspects relevant for models of HBPC and rHBPC
through applying the methods developed in the INTEGRATE-HTA project10 for the socio-cultural
assessment by using a participatory approach.
Methods
The guidance for the assessment of socio-cultural aspects (Mozygemba et al., 2015) offers a five-
step-process to assess socio-cultural aspects in HTA (see Figure 3.6). It also provides a framework
(Figure 3.7) that can be applied on step 1 to 4 of the assessment process. The categories of that
framework resulted of a systematic literature search. Studies referring to socio-cultural aspects of
health technologies were identified and analysed. As presented in figure 3.7 the framework consists
of 11 categories (3 main categories and 8 sub-categories). Cultural Theory11 was used to capture
heterogeneous perspectives related to rHBPC. The application of Cultural Theory requires the
researcher to engage in theoretical reflection as part of all of the steps, although this is most evident
in step 3.
9 Socio-cultural aspects include topics, themes, issues, subjects as well as their features, characteristics and traits.
10 Lysdahl et al. (2016a) 'Guidance for assessing effectiveness, economic aspects, ethical aspects, socio-cultural aspects and legal
aspects in complex technologies.
11 For capturing cultural heterogeneity we used Cultural Theory as an example. Cultural Theory is a common approach in political
science, which identifies four cultural types that differ in their way of organizing social relationships. These are the hierarchical culture, the individualist culture, the egalitarian culture, and the fatalist culture. To capture heterogeneity, we reflected on each framework category against the background of each cultural type. For more information please see the guidance for the assessment of socio-cultural aspects (Mozygemba et al. 2016).
90
Figure 3.6 Assessment process for the assessment of socio-cultural aspects
(Mozygemba et al., 2016)
Kristin
1
HTA commissioners’: a) integration perspective,
b) appraisal vs. assessment aims
Shared objectives with socio-cultural and/or
legal approaches
0. Assess complexity of the intervention (together
with ethical aspects)
1. Identification and prioritization of relevant aspects and stakeholders
2. Validation of identified aspects
3. Assessment of prioritized aspects
4. Presentation of evidence
Collecting intervention information from
stakeholders (SAPs), internet, literature,
umbrella organizations etc.
HTA/ methods contextSteps in socio-cultural assessment process
Intervention context
Stakeholder information
Stakeholder can beinvolved
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Figure 3.7 Overview of the socio-cultural aspects represented in the socio-cultural framework
(Mozygemba et al., 2016)
Socio-cultural aspects identified as important for
HTA in general
Social construction/
understanding of
health issue
Social image/
understanding of
technology and use
Socio-cultural aspects of
technology implementation &
organization of use
Perceived
usefulness and the
idea of benefit
Knowledge
about and
understanding
of technology
Attitudes and
acceptance of
technology and
use
Risk
perception
and
handling
Socio-cultural
aspects of
target groups
Social
inequality and
technology use
User-
professional
relations and
decision-making
Relations between
professionals
providing the
technology
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A total of three, audio recorded, advisory consultations were conducted with nine stakeholders in England
either face to face (n=4 lay stakeholders), by telephone (n=2 professional stakeholders) or using video
Skype (n=3 professional stakeholders). Stakeholders were not required to complete any tasks prior to the
meeting. However, to reassure stakeholders about the content of the meeting, the coordinator of the
stakeholder advisory panels (SAPs) in England sent information about the project along with the
questions to be addressed during the meeting prior to the consultations via email. Both the person
coordinating the SAPs in England (a native English speaker) and the researcher applying the methods (a
non-native English speaker) were present in all of the SAP meetings conducted to test the socio-cultural
assessment process. At the start of each meeting, we defined reinforced and non-reinforced models of
home based palliative care again verbally and (in all group meetings) also in writing on flip chart. We
asked for the experiences of stakeholders in HBPC and rHBPC explicitly at the beginning of the
consultation.
The following paragraphs summarise the five steps of the socio-cultural assessment process (see Figure
3.6) and its application in the case study on models of (r)HBPC. Please find a detailed description of the
five assessment steps in Mozygemba et al. (2015).
Step 0: Assess the complexity of the intervention
The complexity of HBPC was assessed through the application of the five complexity components (i.e.
variety of perspectives, indeterminate phenomena, uncertain causality, unpredictable outcomes, and
ethical complexity12) presented in the ethical domain (see appendix 29).
Step 1: Identification and prioritization of relevant aspects and stakeholders
Step 1 aimed to identify stakeholders and socio-cultural aspects relevant to (r)HBPC and to capture the
heterogeneity of the field. Step 1 consists of three sub-steps 1a, 1b and 1c.
Step 1a: First overview of aspects and identification of relevant stakeholders
Individual consultations lasting about an hour took place via telephone with two ‘pre-informers’ (i.e.
experienced professional palliative care stakeholders) in England to identify socio-cultural aspects
relevant to (r)HBPC and stakeholders that could be involved in the assessment. Both advisors were very
experienced palliative care practitioners. One had excellent local knowledge in practice and the other had
a range of practice experience but is also an established academic able to provide an international
perspective and to draw comparisons between countries. The questions were open in structure and
developed with reference to the socio-cultural framework (see Appendix 30 for more details about the
stakeholders involved and interview guide)). At the beginning the stakeholders were asked to provide
their understanding of HBPC and rHBPC. Both pre-informers were able to suggest stakeholders that
should be involved in the assessment.
Step 1b: Elaboration on identified aspects and first prioritization
Step 1b would have been to consult stakeholders identified in step 1a individually, using the socio-cultural
framework as a structure. Step 1b aims at an individual elaboration and prioritisation of the socio-cultural
aspects identified. However, the logistics of organising meetings with the suggested stakeholders in the
time available did not allow the inclusion of these stakeholders as well as to conduct step 1b.
12
Lysdahl, KB, Mozygemba, K, Burns J et al (Eds) (2015) 'Guidance for assessing effectiveness, economic aspects, ethical aspects, socio-cultural aspects and legal aspects in complex technologies.
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Step 1c: Group consultations to ensure heterogeneity in different stakeholder groups
The first two hour group consultation took place face-to-face with four lay stakeholders (all were relatives
of patients with different experiences in the field of (r)HBPC). Consultation was conducted using an open
question guide developed on the basis of the socio-cultural framework (see appendix 30 for more details
about the stakeholders involved and interview guide) and the information collected through the pre-
informer consultations.
At the end of the meeting, a prioritisation exercise took place. In this exercise, the aspects identified by
the pre-informers were presented on moderation cards. Stakeholders were asked to add missing
aspects. Stakeholders were then asked to prioritise the aspects identified in order of importance for
HBPC by awarding 10 points to one or more aspects of their choice (i.e. all 10 points could be awarded to
one aspect or the 10 points could be allocated to more than one aspect)13. The prioritised aspects were
clustered again and the socio-cultural aspect with the most points was identified – i.e. ‘the relationship
between users and professionals in HBPC’. All aspects that arose in discussions were subsequently
clustered using the socio-cultural framework.
Step 2: Validation of the identified assessment steps
The aim of step 2 is the validation of the aspects prioritised in step 1. The prioritization of the most
important aspects/ framework category will be validated. Differences between stakeholders and
stakeholder groups and changes of stakeholders’ priorities become transparent and can be reflected on.
Aspects identified as being most important for the HTA will be evaluated in the next step of the
assessment process. Due to the lack of heterogeneity in stakeholders and the organisational restrictions
in the case study we decided not to use additional validation. Instead, we focused on the application of
the socio-cultural framework in step 3 only.
Step 3: Assessment of prioritised aspects
Three professionals were consulted in a group via Skype about the socio-cultural aspect identified as
most important in step 1c. The objective of step 3 was to gain a deeper understanding of that aspect (i.e.
“the user-professional-relationship and decision making”). To elaborate on that aspect we applied the
questions offered in the socio-cultural framework (see Mozygemba et al, 2015). A reflection on this
aspect by the application of Cultural Theory (see Mozygemba et al, 2015,) was included in step 3. The
questions and a typology of the four different cultural perspectives (identified by applying Cultural Theory)
on the identified aspect were shared with the stakeholders prior to the meeting (see appendix 31 for more
details about the stakeholders involved and interview guide)).
A participatory approach was used primarily to test if stakeholders could apply the typology developed in
the framework within the consultation. The coordinator and three professionals came together for the
group consultation in England which was conducted via video-Skype by the German researcher applying
the socio-cultural framework. Prior to the meeting, documents about the content of the consultation
(including the framework to be discussed) were shared. After the meeting, relevant information from step
3 was applied to other categories of the socio-cultural framework as appropriate by the researcher.
Step 4: Presentation of evidence
A description of all categories of the socio-cultural framework (see Figure 3.5) is presented in the
methodological guidance for the assessment of socio-cultural aspects (Mozygemba et al, 2015). The
evidence collected through the whole assessment process was structured using the socio-cultural
framework as a template. Information on all framework categories is provided within the “findings” below.
13
Results of the prioritization exercise conducted in step 1 using the group of relatives are presented in appendix 32 of the case study.
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Findings
The information presented here is based on the stakeholder consultations, which were conducted with
the objective to apply the developed assessment process and the socio-cultural framework. Due to the
homogenous stakeholder sample and the organizational necessity to fulfil different assessment steps
with one stakeholder group only, results do not represent the variety of the field. However, they show how
various socio-cultural aspects of rHBPC can be identified and assessed by using the socio-cultural
framework and a participatory approach.
The information collected on socio-cultural aspects relevant for assessing (r)HBPC was structured by
applying the socio-cultural framework (Figure 3.6) and is presented in the following part of the report. All
framework categories were addressed. To allow comprehensive information on each aspect relevant for
more than one category, information collected throughout the whole assessment process was provided in
all relevant categories (see Appendix 32).
Framework category 1: Social construction/ understanding of health issue
Category 1 of the framework refers to the different ideas related to a health issue existing in a society.
The understanding of health issues and related treatment options can differ depending on the socio-
cultural context of people. The way individuals, a group or a whole society defines/perceives a health
issue influences how and for what purpose a technology will be developed, assessed, used, and
implemented. Technologies in themselves can also change the way in which health issues are socially
defined.
In HBPC the health issue is understood as a “journey”: HBPC characterizes the health issue as a
“journey” (the dying process) which includes the family as a unit This also includes informal caregivers
getting support from HBPC by themselves (e.g., after the patient died) and health issues related with
overtaking the role of a caregiver. The “journey” consists of phases characterised by changing support
needs of patients and families/ informal carers. This is linked with uncertainties regarding starting and
transition points of the “journey”. Professionals guide through the phases, preparing and advising
(unexperienced) patients and relatives. This approach is different to curative health services, which
focuses on specific diagnosis and related treatment options. The unspecific focus “end of life care” can
lead to uncertainties e.g. concerning access to rHBPC and HBPC.
The health issue in HBPC is seen from a holistic perspective: In HBPC, patients define their health issues
(relatives and professionals are indirectly involved). For example a GP’s referral to palliative care can be
reasoned in the reduction of physical pain, which fits the system where diagnosis and treatment are
assessed by a health expert. Entering HBPC can question the medical appraisal, e.g. through asking if
pain reduction is the patient’s main problem. Although diagnosis can determine access to HBPC, it is not
emphasised as central for the understanding of the health issue. HBPC holistically addresses physical,
emotional, psychological and social needs of the patient (e.g. pain understood as bio-psycho-social
phenomenon, cannot only be treated with pain killers). A reliance on solely medical interventions for pain
management is not appropriate within HBPC as all elements of ‘pain’ must be addressed.
Multi-professional perspectives emphasise health issues differently in HBPC: Multidisciplinary teams are
involved in HBPC to ensure a holistic perspective on the health issue. This also leads to different
understandings of health issues and appropriate treatment foci on professionals’ side.
Medicalization of dying processes: Issues of professionalization and medicalization of dying processes
were mentioned. However, stakeholders argued that following a patient-centred approach, not defining
health needs by professionals or informal carers, might limit medicalisation.
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Framework category 2: Social image of technology and use
Technologies and ideas of health and illness are mutually linked social constructions, produced and
confirmed by social actions. As such, a technology is influenced by cultural and social norms, values and
expectations linked to the health issue and vice versa. These values, norms, expectations influence how,
when and for what purposes a technology is designed and implemented.
Associations with HBPC: A focus on the end of life frames HBPC in England. Patients’ and relatives’
ideas of death and dying influence what they expect of HBPC. E.g. being referred to HBPC, which is
culturally associated with a close death, could challenge patients’ and relatives’ quality of life. Knowledge
about HBPC-services and the way they support a good quality of life in the last days could change
patients’ and relatives’ picture of the intervention as well as expectations related with HBPC.
Professionals will elaborate on the idea of HBPC in the individual case together with patients and
relatives.
Home as the best place to die: HBPC is linked to the idea that “home is always the best place to die”. But
at least the patient-centred approach, requesting decisions on individual cases’ base, relativises that
generalisation. The idea of dying at home as central for HBPC was associated with benefits for patients
and relatives being familiar with the situation at home. But apart from the changes the familiar situation
will be passed due to the implementation of HBPC, concerns were expressed about the potential of
(economic) misuse if people are pressured to die at home. These concerns were based on the
assumption that hospital care or hospice care would be more expensive than HBPC – e.g., given the
number of unpaid informal caregivers involved in HBPC.
Informal caregivers are part of HBPC: HBPC addresses the family as a unit of care – taking health needs
of patients and relatives into account. Related with the involvement of informal (unpaid) carers there is a
political image of HBPC as charity (“26,000 unpaid carers and they are doing a 90 hours week”), which is
linked to an image of informal carers as heroes. This position of informal caregivers as main carers in
HBPC was described as politically controversial. Questions of funding issues are related. In parallel,
caregivers are seen as vulnerable group due to potential health risks related with their burden of care.
Health risks become especially important against the background of socio-demographic challenges such
as an ageing population: In the future more very old and non-healthy carers will take over the caring role.
Related are questions for additional caregiver support, which could be a benefit of rHBPC.
HBPC challenges the structures of the health system: Patient-centeredness in HBPC focuses on
individuals. This could challenge the solely medical approach – a main organizational characteristic of the
health system. The medical approach is characterised by clearly defined health issues (diagnosis) and
treatment options chosen by a medical expert. HBPC however, requests flexible and patient-centred
needs assessments and focusses “on services that assist the person, not vice versa”. Challenges related
to these characteristics of HBPC could cause changes in HBPC (e.g. increase of professional authority),
but also changes of the system (e.g. decrease of hierarchical organisation in multi-professional teams).
HBPC services are various and continually developing. This is linked to insecurity about the continuity of
care regarding patients, relatives and professional providers. The variety of services also combines
different approaches/ cultures of care. As an example stakeholders mentioned difficulties to combine
clinical health care and community health care as well as social care.
Framework subcategory 2.1: Perceived usefulness and the idea of benefit
The category “perceived usefulness and the idea of benefit “refers to the evaluation of a technology.
Understanding and evaluation of a technology are socially and culturally influenced. This includes various
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preferences for outcome measures and differences in the understanding of effectiveness and safety.
Furthermore, HTA as an assessment procedure is itself culturally shaped.
Benefits for patients and relatives: HBPC addresses the family as a unit. Consequently benefits need to
be described for patients and informal caregivers as well as for professional providers. Questions how,
when, and how often needs assessments should take place are related. Being in a familiar environment,
feeling safe and secure is seen as benefit for patients. It is assumed that HBPC creates normality and
autonomy as it would not be possible in care institutions. A flexible response to patients’ needs at the end
of life is difficult to provide in a clinical setting. Relatives can be closer to their loved ones (e.g., no need
to rush into a hospital) and manage their own life easier (e.g. fulfil other family duties etc.). HBPC
incorporates relatives as part of the patients’ “journey” and may continue through the bereavement
process. Support services addressing relatives explicitly (e.g. a sitting service that allows for off-time or
social activities to protect caring relatives for social isolation) are part of HBPC - sometimes as rHBPC.
However, empowering relatives could decrease the patient’s benefit, e.g. if relatives decide to give up the
role of being an informal carer.
Benefit for professionals is seen as the chance to deliver care on a very individual basis, which is both:
challenging and stimulating. They bring the professional skills understood to serve needs expressed by
patients and relatives. The cooperation of different services, team work, shared responsibilities, etc. could
also result in benefits if clear pathways are provided. Cooperation between community and clinical health
care providers seems to be (culturally) challenging. It was stated, that HBPC professionals should have
additional skills in community care and it was stated that they are often not trained enough. Differences in
professionals’ HBPC-training can also affect the benefit of HBPC directly (dealing with the patient) and
indirectly (working together with professionals who have different standards).
Benefit assessment of HBPC: The variety of perspectives involved in HBPC (e.g., different professional
cultures, heterogeneity of patients’ preferences) also challenges the way benefit is defined/ understood.
Furthermore the understanding of HBPC can differ between (different) service providers and decision
makers (e.g., focus on pain killers or psychosocial aspects of pain).
Framework subcategory 2.2: Knowledge about and understanding of technology
What is considered as “the right” knowledge and how it is legitimised is socially negotiated and can differ
between groups. Knowledge about a particular technology influences its social images. Understanding
the purpose of a technology basically shapes decision-making at the political level as well as in concrete
treatment situations. This framework category encompasses the value that is assigned to new
technologies and their implementation, user ideas, ideas that influenced the technologies’ development,
and knowledge about treatment alternatives in comparison to the technology of interest.
Understanding and expectations of patients and relatives related with HBPC: Knowledge and
understanding are keys for being confident with the services and the situation in HBPC. HBPC and
rHBPC contain a broad variety of services, which are often not known by patients and informal caregivers
and which differ regionally. Additionally expectations of patients and relatives related with HBPC can be
very different. Professionals should continuously obtain a common understanding and inform about
available services from the very beginning. It is assumed that patients and relatives ask more about
services they know than about services in HBPC in general.
Choose the right contact person: The variety of services and providers involved in HBPC lead to
questions of service coordination. E.g., GPs as well as informal caregivers can be the coordinator.
Related are questions of (professional) responsibility, access to information about services and funding
opportunities (e.g., informal caregivers who took over the role as coordinator reported about “battles” to
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get information and funding, which negatively influenced their own health). The role of community nurses
in coordinating care was emphasised, but they are not perceived as coordinators in HBPC.
Framework subcategory 2.3: Attitudes and acceptance of technology and use
An attitude is a summative assessment of a person, group, or the public assessing the technology at a
certain point in time. Attitudes are related to beliefs about science, social trust, perceptions of benefit and
risks of a technology. They express fears, hope, curiosity, and security ideas that are linked to the
technology of interest. Attitudes towards a technology characterise its social image. They are influenced
by knowledge about the technology, the perceived usefulness as well as by socio-demographic variables
such as gender and age. They are also part of the social and cultural context of users, providers and
decision makers. Refusal and acceptance of a technology are consequences of attitudes associated with
a technology.
HBPC is based on societies’ ideas about accompanying dying people during their last journey.
Professionalisation in HBPC will influence these ideas and vice versa the ideas will change HBPC. The
symbolic value of HBPC for patients and caregivers correlates with the acceptance of the care situation
by family and friends. The cultural context shapes HBPC (e.g., attitudes to HBPC of people living in an
area with strong social networks and social support will be different to attitudes of people living in an area
characterized by anonymity and individuality). The cultural background also influences role expectations
(of being a patient or an informal caregiver). E.g., an area valuing family support high could socially
require taking over the role of an informal caregiver as a duty, especially for women). (r)HBPC can
change these perspectives e.g. through the empowerment of informal caregivers. However, this can also
challenge the informal carer’s position as a member of the given social environment. For instance, the
question whether it is acceptable to stop caring depends on the expectations regarding the informal
caregiver role.
Attitudes towards HBPC are influenced by preconceived ideas of the purpose of HBPC and related health
services. For example, expectations to HBPC might differ, if informal caregivers understand professional
services as support in fulfilling their role of being an informal caregiver or if they see them as the
responsible providers. But also attitudes to the special services itself can influence the acceptance of
HBPC (e.g., a general suspicion about social care related with support of disadvantaged groups).
Another background idea related to HBPC is the attitude that “dying at home is the best option for
patients”. This is related to a view on HBPC as charity, which leads to controversial discussions about
the provision of financial resources. In addition, views of public and policy makers may differ to views on
organizational level.
Framework subcategory 2.4: Risk perception and handling
“Risk perception and handling” refers to culturally different definitions and perceptions of risk and the
corresponding ways to handle these (Beck, 1992). Risk assessment usually focuses on non-desired
consequences of a technology’s implementation. However, risk can also be seen as a chance for
development.
In HBPC we identified the following risks relevant for the socio-cultural assessment:
There is a risk of pressuring patients to die at home without assessing their individual needs,
because of the belief that home is always best (see the framework category ”attitudes and
acceptance of technology and use”).
HBPC is related to the risk of “hospital at home”. Equipment and the variety of services involved in
HBPC can overwhelm and confuse patients and their families. In addition, the family might
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perceive professionals as strangers in the patient’s home, which could also cause feelings of fear
and mistrust (e.g. that something will be stolen).
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Feasibility of HBPC is associated with the number of agencies involved in the care situation. If
professional providers are not well coordinated HBPC could fail or remain patchwork instead of a
holistic and patient-centred intervention. This includes for example questions of different
professional training, different professional cultures and cooperation styles.
There is a risk of overburden and social isolation for informal carers as well as the risk of injury,
which may occur during wrong handling of the patient. Therefore, professionals recommend that
informal carers need assistance e.g., if own health problems arise. rHBPC may be an option to
support informal caregiver’s health and wellbeing.
Professionals providing HBPC could become too involved in the complex care situation of a
specific family. Accordingly, professionals should explicitly communicate that HBPC is delivered
by a team to patients and families.
A lack of flexibility and a lack of a holistic perspective in the care plan as well as in the needs
assessment can lead to wrong conclusions regarding patients’ needs.
HBPC is described as a developing mode of care. Many projects are field tested and not
established as continuous services. As a consequence, neither professionals providing HBPC nor
patients and relatives can trust that services are continuously available. Due to this insecurity of
services there is a risk of fragmentation and discontinuity of care.
Framework category 3: Socio-cultural aspects of technology implementation & organization of use
“Socio-cultural aspects of implementation” focus on characteristics of the target group, social inequalities,
the relationship between user and professional provider, decision-making in the concrete treatment
situation and relationships between different professionals providing the technology.
In general, the patient-centred approach associated with individuality challenges the health system (with
clearly defined diagnosis and linked (curative) treatment decisions and with a focus on professional
standards). In addition, HBPC includes a variety of services, which are continually developing. This
variety of services combines different approaches/cultures of care, which can cause difficulties e.g. to
combine clinical health, community health, and social care.
The application of the four cultural types resulted in the realization that HBPC seems to be a fast
developing area of services which struggles to bring egalitarian service ideas14, such as shared
professional responsibility and criticism of hierarchical organisation, and hierarchical service organization,
as it is typical for nursing homes or hospitals, together in a care context. The egalitarian idea represented
in palliative care could influence the health system and hierarchical structures, e.g. when HBPC providers
train nurses who are working in a hierarchical context of a nursing home. As well, HBPC might need to be
adapted to established care structures, which could challenge the egalitarian way HBPC is shaped.
14
“Egalitarian ideas” refers to the egalitarian culture, which emphasizes a social equality and denies hierarchy and economic benefits. A more detailed description is provided in the guidance to assess socio-cultural aspects in HTA (Mozygemba et al., 2015).
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Framework subcategory 4.1: Socio-cultural aspects of target group
The characterisation of the target group of the intervention is important in understanding e.g. compliance
and refusal of treatment and how the family is affected by the intervention. Beyond this ideas (inner-
stereotypes) of (future) users influence the design and development of a technology. Target groups are
often characterised by a set of socio-demographic variables such as age, gender, educational level, race,
ethnicity, religion etc. The characterisation by socio-cultural context and taking the social network and
related aspects into account could be advantageous when describing the target group of a technology.
Information about family roles and related expectations, the idea of being a patient, or the way the social
support network works, help to understand whether and why technologies fail, or succeed in a specific
cultural context.
Differences between patients and patients’ families: HBPC is appropriate for patients at the end of life
and their families. In contrast to usual health care, HBPC addresses each patient as an individual.
Patients and their families differ regarding diagnosis, needs and expectations, ideas of being a patient,
ideas of care provided by relatives, ideas of death and dying etc. In addition, social needs can differ from
clinical needs. The amount of social support and the related relief is also different (e.g. relatives could
share the caring role or get additional support from the church). The availability of social support may
also change during the “journey”. The degree of acceptance of the patient and caregiver’s situation by
family and friends will influence the symbolic value of HBPC for patients and caregivers.
Although cultural differences based on ethnicity and religion can be assumed, there is not the diversity of
care experiences in the sample that would allow reporting on that. However, stakeholders express that
community care is more experienced in working with people from diverse cultural backgrounds.
Furthermore, less affluent areas are described as having the greatest family support with regard to the
socio-economic status and the level of education. The formal educational level is more diverse in areas
with less family support. Higher education level often lead to more information requests (asking for
information, doing own research) and higher expectations of the services. Informal care givers tend to be
female.
Patients’ needs are central in HBPC. But as the social environment of the patient the wellbeing of
informal carers becomes also important for HBPC. Due to the socio-demographic change future informal
caregivers will be of older age and having worse health status, which emphasises the question of
caregiver support. However, medical prescriptions for informal carers are critically viewed – e.g. due to
related risks of overtreatment and medicalization. Informal carers’ needs differ (in addition to socio-
demographic variables) with regard to the social roles they have (e.g. as mother caring for children or as
an employee) and their experiences of caring. Informal carers may experience changes of their social
roles, financial income, loss of employment and social status as a result of undertaking HBPC, potentially
becoming patients themselves as a result of the burden experienced through caregiving. Culturally
different are the expectations to take over the care giver role, e.g. feeling a duty to care for a husband vs.
free decision to support a close friend. HBPC can influence that perspectives and vice versa.
The relationship between patients and relatives: Relationship between patients and relatives are
influenced by gender, social power dynamics (e.g. who makes decisions), health status and age of the
carer themselves. HBPC comes with consequences for the whole family related to family dynamics. At
the beginning, the family does often not realise the difficulties and complexity of caring a patient at home.
Difficulties can result from a variety of causes (e.g. a patient’s wish to not involve relatives in care
decisions or the patient’s mental capacity to make decisions). Changing roles in the relationship (e.g. the
role of being an informal carer dominates the role of being a husband) can cause distress. Professional
providers emphasize to remind patients and relatives of their original relationship, e.g. as a wife or a
daughter. Contact to other patients and informal carers, e.g. as part of rHBPC, can help to prevent social
isolation, to exchange experiences and to find new support.
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Framework subcategory 4.2: Social inequality and technology use
A technology’s implementation and use is linked to questions of social inequality. Social inequality refers
to the disparate distribution of material and immaterial resources in a society and its consequences for
social participation. It is linked to issues of stigmatisation and discrimination. The focus is on
disadvantaged groups e.g. in accessing the services. Besides this, discrimination can take place if a
technology addresses its target group as an entity, without considering different needs.
The access to HBPC and rHBPC can become an issue of social inequality, e.g. if services are not known,
e.g., if HBPC is related with specific diagnosis, or if funding information is lacking. Social inequalities can
arise for several groups: ethnic minorities, older adults, patients with non-cancer-diagnosis, prison
populations, those in residential and nursing homes etc. Reasons for differences in access are: 1) cultural
stereotypes (such as “ethnic groups care for their own family at home”), 2) scarcity of providers, 3)
resource constraints, 4) knowledge of services15, 5) local infrastructure, 6) focus on specific diagnosis, 7)
the availability of a social support network, 8) continuity of services (services are not available anymore).
In addition families differ in their ability to deal with stress searching for support, which is also related to
specific competencies. To ensure equity HBPC providers emphasise the strategy to take more time and
resources for disadvantaged people. Beyond the individualised approach of HBPC seems to involve
inequalities of care legitimized in the different individual needs. I.e., equal access to HBPC is not fulfilled
if all have access to all services in general, but if they have access to the services which suit the
individual patient’s needs.
Framework subcategory 4.3: User-professionals-relationships and decision making
Relationships between users and professionals are embedded in an institutional (socio-culturally shaped)
context. These relationships are more or less formally shaped and, include more or less prescribed social
roles linked to different expectations of autonomy and responsibility. Professionals and professional
cultures involved in treatment and decision processes should be described in the socio-cultural
assessment. Mutual expectations will influence the way treatment is provided. For instance, ideas of
authority and autonomy have to be in accordance with each other to ensure successful treatment. Users
and providers socially negotiate to see if their ideas are compatible or if the socio-cultural (institutional,
respectively) context of treatment needs to be changed.
The relationship between professional service providers and patients: Relationships between
professionals and service users in HBPC often differ from usual health care. They differ with regard to
the idea of being a multidisciplinary egalitarian team providing patient-centred and holistic care,
which takes the family as a unit into account,
the higher degree of intimacy between professionals and service users,
the limited period of time HBPC is provided,
a wider range of involved professional services,
the necessity to combine social, clinical and community health care,
the location of care, i.e. the patient’s home.
Patient-centeredness in the user-professional-relationship in HBPC: Patient-centeredness in HBPC
means to focus on the needs expressed by patients (and if necessary, needs indirectly assessed by
15
The knowledge about services differs due to users’ educational level and health status. Users also often use the internet for information.
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relatives or professionals (perceived needs)). Patient autonomy and shared decision making are
emphasised in HBPC.
The application of the four cultural types to this framework category (Mozygemba et al, 2015) shows that
patient-centeredness – despite the egalitarian ideal of HBPC – results in relationships that contain a
mixture of all four types (see Appendix 33). Depending on individual needs their changes during the
process of dying, professionals can immingle between relationships that are more individualistic,
hierarchical, egalitarian, or fatalistic. E.g., sometimes (at the beginning) patients can be very fatalistic, not
believing that they can shape anything.
Then they gain more information and gain confidence about managing their own situation and move to a
more individualistic approach (e.g., requesting special HBPC services) or they may have been quiet
egalitarian (e.g., meeting their professional carers as equals and engaging in shared decision making)
until they become quiet medically unwell (which could for example result in the request for authority).
Central for professional providers seems to be an egalitarian, democratic ideal of the relationship
combined with an emphasis of individualism of the patient and the family. The overall aim is to care for
the patient empathetically - led by the egalitarian team ideal. A hierarchical structure was explicitly
declined by HBPC-professionals. It seems to be only acceptable if it is related to a patient’s individual
request of authority. This culture of HBPC can be challenged when it is provided in a hierarchical
organisation such as a nursing home, but also in a hierarchical organized family system.
A patient centred relationship demands the adaptation of the services to the patient and not the other
way round. Needs assessments should be a two way procedure based on talks between patients and
professionals. To ensure a realistic mutual picture of the services professionals inform the patient about
HBPC and listen to their expectations at the beginning of HBPC. The first visit is the basis for establishing
a confidential relationship. The lack of a standardised frame shaping that relationship is of advantage
(more freedom and individuality), but also challenges professional care provision. Furthermore, patients
are from day to day different with respect to their needs and expectations. If care providers notice that the
patient is not well and cannot work with the care plan they have to adapt it in the sense of the patient.
Conflicts can arise if patient’s expectations do not fit with the way professionals would deal, act, and
advice or if patients/relatives are not convinced that professionals act in their needs. E.g., patients could
wish not to involve relatives to know everything about their condition, his/her choices, and wishes.
Although professional providers will advise the involvement of relatives in decision-making, they have to
respect patient’s choice. Difficulties can also arise if users do not get along with their care providers.
Consequently, other professional carers have to be found. Another limitation to fulfil patient’s preferences
results from (staff) resources of HBPC, e.g. if there is a number of individual patients who want to get up
at 7 o’clock.
Relationships between the HBPC-team and service users should be stable and continuous. The GP is
seen as “critical link” in the provision of HBPC-services. Problems may arise when care is delivered by
large teams. The commissioning of additional care providers from different agencies (stakeholders
mentioned that 20 professional carers a week is not unusual) can cause an unsettling feeling for patients
and informal carers. The situation also challenges providers not specialised in HBPC (e.g., the intensive
nursing service, which is not specialized in psychosocial support but integrated in providing care in HBPC
and challenged by the relatives’ wish for psychological support). Empowerment in that intensive – non-
holistically operating – service is more viewed in training relatives in medically dealing with symptoms,
medication etc.
Degree of intimacy between professionals and users: Due to the advanced dying process, HBPC patients
and relatives cannot become familiar with the processes of HBPC in the health system. Professionals
function as empathetic gatekeepers to services and equipment.
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Patients and relatives also often share their stories with the professionals - not only their physical
symptoms. This bases a degree of intimacy which contrasts e.g. the hospital setting, where it is unusual
for such relationships to develop.
The user-professional-relationship characterises intensive contact and communication. However, the
closeness in the relationship can be misunderstood by patients and relatives as a kind of friendship and it
can result in a burden for professionals if they become too personally involved in that complex care
situation.
The relationships between users and different professionals in HBPC: HBPC is delivered by a multi-
professional team. Patients and relatives should know whom to contact for what purpose and when.
When this happens successfully, it gives a feeling of confidence and trust to service users. But often
there are several service providers and different agencies involved in HBPC, which causes different
degrees of intimacy in the relationships to different providers. Different professionals will also shape the
relationships differently due to their professional focus and culture.
E.g., there are differences between the patient-professional-relation in community care and health care.
In addition stereotypes linked to specific professions can influence the relationship, e.g. service users
being suspicious of social workers because they are seen as professionals working with disadvantaged
people.
The relationships between professional service providers and informal caregivers in HBPC: Informal care
givers are part of the holistic approach of HBPC, but professionals need the patient’s permission to
involve them in the care. Professionals see informal carers as 1) co-client (resource of patient support),
2) co-worker (wellbeing important for continuing care), and 3) superseded carer (not needed if
professionals are involved).
1. Informal carers as co-client: HPBC is linked with a change of family roles and relationships. The
patient and relatives become a care-recipient with the related changes in their social roles and
networks. Informal caregivers have a special role in HBPC: As persons involved in the journey
and confronted with the burden of care HBPC addresses caregivers as a vulnerable group. Within
this context rHBPC is of interest, because it aims to promote the wellbeing of informal caregivers.
HBPC supports informal carers for example through facilitating the exchange with others involved
in similar situations, through training in handling the patient as well as through psychosocial
empowerment e.g. in reflecting on the role of being an informal carer. None the less, it is politically
controversial discussed if informal carers are seen as vulnerable targets of HBPC with needs that
have to be addressed alongside the patient’s needs. Furthermore, carers are not used to
expressing their own needs and often need to be asked to do so.
2. Informal carer as “main carer” or “co-worker”: On the other hand informal carers become the
“main carer” collaborating with the HBPC team. E.g., they coordinate care, interface between
professional care and the patient, and are involved in handling, mobilising and medication, etc.
rHBPC aims at empowering informal carers, e.g. through training in handling the patient or in
reflecting about their own needs. This support could change the way professionals see informal
carers. However, empowering informal carers can also change the way informal carers value the
caring role and the related burden.
Decision making in HBPC: Professional stakeholders emphasized that HBPC aims at informed consent
and shared decisions in all stages of the treatment process. It should be based on confidence and
continuous communication between professionals and patients. Patients and informal carers benefit from
a continuous caring relationship with the HBPC team. “Knowing who to contact when” improves
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confidence and safety and prevents revolving door effects, i.e. patients who often contact emergency
services.
Short distances and intensive communication lead to quick support and to referrals to the “right” places.
Patients and informal carers need to be fully aware of the patient’s prognosis (not only death, but the
process of dying16) to make an informed decision about using HBPC. Professionals have to be sensitive
to the verbal and non-verbal cues of patients and informal carers indicating their readiness to deal with
discussions about dying. Decisions are prepared in the team of providers, sometimes in joint meetings
involving patients and relatives. Professionals can try to structure a care plan in family meetings – “having
a cup of tea together with the family”.
Shared decision making in HBPC has to ask 1) if mutual discussion between patients and (informal)
carers are always taken into account and 2) if attention to the patient’s perspective/requests is possible in
providing the services. It needs 3) to be flexible, because of changing patient’s needs/ informal carer’s
needs or because of the various stakeholders involved (coordination of a team working together vs.
independent organisations for different services, decisions made for an individual will affect the whole
family). 4) How does the patient/informal carer understand the information provided by HBPC-
professionals and what do they expect? The culture of decision making influences if patients and
caregivers are used to making their own decisions or if they wish for ownership over their own care plan.
In addition professionals experience difficulties when applying the patient-centred approach due to their
own professional socialisation in being an expert which places them as leaders in the decision-making
process. A hierarchical institutional context (e.g. in a nursing home) can challenge autonomous decision
making of patients.
Upholding patient wishes may lead to potential conflicts with professionals own beliefs or decisions. The
amount of information for informed consent on the treatment options will vary according to individual
circumstances. Contradicting wishes of patients and informal carers can result in a conflict of interest
between patients and carers (e.g. the patient may prefer to be cared for at home, but the informal carer
may prefer care to be provided in an institution). Furthermore key issues of concern around informal
caregiving lie in ensuring that caregivers are willing (not coerced) to undertake the caregiving role and to
make an informed decision about adopting the role. Informal caregivers may feel morally obliged to take
on and/ or continue caregiving as a result of promises made to the patient (e.g. to fulfil wedding vows or
promises made during the illness period). It needs also to be ensured that they have the ability to provide
care at home. Increased vulnerability of patients and informal carers (e.g. due to patient capacity,
physician/ staff paternalism, or restrictions on choices available) can also challenge shared decisions.
The issue of responsibility in HBPC: Issues of responsibility in HBPC were reported. Clearly defined
pathways indicating who is ultimately responsible for relevant decisions are missing. The patients’ wish
for control challenges the question of responsibility for care in addition – even more if there are issues of
the patient’s mental capacity. Responsibility for care decisions can be also challenged by cultural
different perspectives on the way decisions should be made. From the application of the cultural types it
becomes visible, that professional care experts have the responsibility for care in a hierarchical setting
(e.g. in a hospital). This is linked to a decreased patient autonomy. Egalitarians emphasise a democratic
relationship between patients and professional providers. This includes different views on decision
making and responsibilities compared to a hierarchical context. E.g., responsibility in an egalitarian
setting is shared within a team of professionals, or responsibility is shared with the patient and informal
carers. Responsibilities in the egalitarian context seem to be less clear than in the hierarchical context,
which could request negotiation. Interestingly, in the case study the question of responsibility was clearly
16
HBPC pprofessionals are experienced with the different stages of the process of dying. They aim at guiding (more or less unexperienced) relatives and patients through this process and to make the process part of their life - not a „diagnosis of death“ only.
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raised by pre-informers, which had experiences in providing HBPC and by relatives, but the group of
professional providers did not see a problem.
They referred to the GP as the responsible person, which could be a reference to a hierarchical culture.
In parallel this group denied hierarchy as the cultural principle of social organisation in HBPC. From the
socio-cultural perspective it is an interesting question how the social and cultural organisation of
responsibility in that complex situation is solved and how they influence each other.
Framework subcategory 4.4: Relationships between professionals using the technology
Relationships between different professionals providing a particular technology or deciding about it are
embedded in a socio-cultural context. This becomes visible in different working cultures, professional
cultures, ideas of (interdisciplinary) cooperation, questions of social status and related responsibilities,
mutual expectations, as well as in issues of social power in treatment decisions etc. Perceptions of the
health issue and of the technology are influenced by the service culture represented.
Cooperation in a multi-professional context: HBPC is delivered by a multi-professional team. The number
of professionals involved in HBPC varies (due to resources, availability, service model, number of
different agencies involved, etc.). HBPC providers are also gatekeepers to other services (such as
physiotherapy, intensive nursing, psychosocial support etc.). These providers and agencies (with different
(educational) standards and organisational cultures) can be involved in addition and increase the number
of professional providers involved.
Professionals emphasised the importance of multi-professional cooperation in HBPC. Cultures of
cooperation in HBPC-teams differ due to the number of professionals, the variety of disciplines and
professional cultures involved. Challenges are also linked with the necessity to combine health care and
social care, and community health care. Community care competence is seen as base for HBPC, but
stakeholders stated that HBPC-providers are often not experienced enough in that field. An
understanding of community services and palliative care services in comparison as well as an
understanding of potential linkages is important for mutual expectations linked with HBPC. Additionally,
the involvement of informal carers as non-professional carers, but co-workers request reflection on the
way the relationships between them and professionals should be shaped (e.g. in terms of responsibilities
and decision making).
Responsibilities and professional culture in HBPC: Professionals involved in HBPC characterize their
relationships in HBPC-teams as egalitarian and democratic. They explicitly criticise hierarchical structures
and authoritative decision making processes. However, different organisational cultures can challenge/
question the egalitarian approach as well as the hierarchical context in which health professionals are
usually socialized, can influence actions unconsciously (e.g. if the GP is seen as the person responsible
for HBPC). Additionally, responsibility issues can be solved differently in community and medical care.
Questions of responsibility will also relate to the needs assessment. The different ways of specific
professions to understand and to deal with health issues (e.g. focusing on reduction of physical pain
using pain killers as opposed to addressing psychosocial aspects of pain) can be challenging for
providing HBPC.
Reflections
Complexity
Instead of emphasising the assessment of complex technologies, the socio-cultural guidance
concentrates on complexity characteristics of health technologies, assuming that nearly all health
interventions can become complex. The central question for the socio-cultural assessment is whether the
complexity of a technology needs to be considered in the assessment. Therefore the identified complexity
characteristics presented in the guidance on the assessment of ethical aspects (Bakke Lysdahl et al,
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2015) can be also applied (step 0, Figure 3.6). The application of the complexity characteristics in
(r)HBPC revealed a range of features of the intervention that illustrate its complexity as summarised in
Appendix 24.
Integration
A number of common topics, at least relevant for ethical and socio-cultural aspects, can be found in
(r)HBPC. Examples are ‘the user-professional-relationship and shared decision making’, changing social
roles, social inequalities related to access and availability of (r)HBPC. Integration of socio-cultural,
ethical, and legal aspects is closely linked. Integration of effectiveness and economic aspects is still
challenging. Collaboration across all assessment aspects, e.g. in conducting a common scoping
exercise, and through sharing and discussing assessment results will improve integration.
Stakeholder input
Stakeholder involvement is, to our knowledge, the best opportunity to capture socio-cultural
heterogeneity when assessing a health technology. The bigger the variety of perspectives included the
more likely the cultural diversity of a society is reflected. Stakeholders can be involved at all stages of the
socio-cultural assessment process. Their perspectives can help identifying evidence gaps and non-
popular positions as well as reasons for non-acceptance and failure of a specific technology (e.g.,
Reuzel, 2004).
In the case study on (r)HBPC we involved stakeholders to identify heterogeneous stakeholders as well as
to identify and prioritise socio-cultural aspects related to (r)HBPC. Stakeholders were also included to
assess the prioritised aspect – the “user-professional-relationship and decision making” – in detail by
applying the socio-cultural framework and the four cultural types offered by Cultural Theory. Stakeholders
were successfully involved in three ways: Consultations take place individually via telephone, in a group
discussion face-to-face and via Skype. Stakeholders found no difficulties in taking part in the consultation
and felt that their participation was useful, suggesting that the project had a positive impact (see
Appendix 34).
However, as this was a ‘demonstration-HTA’ testing new methods with organizational limitations (e.g.
very limited time for the application of all methods developed throughout the INTEGRATE-HTA project)
and it was not feasible to include a wide variety of stakeholders. We also used the group of relatives to
test the methods of step 1 and the group of professionals was used to test step 3. Ideally, both should
have been part of step 1 to ensure heterogeneity of stakeholder input. The case study results emphasize
the importance of a) the identification and prioritisation of (culturally) different perspectives and b) the
stepwise inclusion of heterogeneity in the assessment process.
In our exemplar application, all categories of the socio-cultural framework were reflected from the
perspective of Cultural Theory. The detailed assessment of the prioritised aspect “user-professional-
relationships and decision making” was supported by a tool providing information on the four cultural
types (see Appendix 33). Stakeholders (not familiar with the application of theoretical approaches)
successfully applied this tool. It was feasible for their experiences about user-professional relations and
decision making in (r)HBPC to be assigned to the different cultural types. This task reflects different
constellations and controversies related with the prioritised aspect. Heterogeneity of perspectives can be
gained by applying the tool, although we had a homogenous group of professional providers. A practical
limitation was related to the preparation of the group meeting17, which could have been improved by
providing a better introduction of the purpose of the tool to participants before the meeting via email.
17
One of the consultants felt like being asked for a test when she got the information about the cultural perspectives prior to the meeting. She felt more confident when she heard what will happen.
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We introduced the intervention and comparator by email and again in the consultation. We explicitly
asked for the experiences in HBPC and rHBPC at the beginning of the consultations. During this process
and the application of the methods, we recognised different understandings between stakeholders
regarding HBPC and rHBPC.
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Several problems were identified: a) the term rHBPC was taken from a literature review on HBPC and not
part of the HBPC-practice, b) HBPC itself turned out to be diverse due to the variety of services it can
include, and c) relative support can be part of regular HBPC. Consequently, the results presented in the
socio-cultural assessment cannot definitively separate the results between comparator and intervention.
We recommend asking stakeholders for their understanding of the intervention at the start of the
assessment and to ask what will be missing if as in our case additional stakeholder support will not be
offered. It could be good to do that individually to avoid any effects of group dynamics.
Due to the difficulties known to exist with priority setting in HTAs, the prioritizing process is separated into
different steps of the socio-cultural assessment process. Again organisational limitations allowed
prioritization only as part of step 1c. The prioritised aspect (“the user-professional relationship and
decision making” is not validated through the socio-cultural assessment – neither was it confirmed by the
same stakeholders after the meeting, nor in a group discussion of different stakeholders. The method of
prioritizing different aspects itself did not cause problems for the participants. However, it could be difficult
to reflect ad hoc on the socio-cultural importance when aspects are added (e.g. if the aspects are more of
economic nature).
For increased understanding, stakeholders should be asked to explain their reasons for priority choices.
Time is an essential issue for that process and needs to be taken into account during preparation for the
consultations.
Value and application of methods
The guidance on socio-cultural aspects was applied in HBPC and rHBPC with a focus on methods
testing. The results of the application provide information about important socio-cultural aspects in
rHBPC, but are not valid enough to ensure a strong statement about rHBPC. Both tools (the assessment
process and the socio-cultural framework) worked well, as far as we were able to apply them, and could
be developed further on the basis of the case study application. Main limitations result from the points
mentioned under “stakeholder involvement” and from organisational issues, which did not allow each
assessment step to be completed as prescribed. Nevertheless, a broad variety of aspects could be
identified.
Overlaps exist with the results from the qualitative systematic review of (r)HBPC and also with the results
of the ethical assessment of (r)HBPC. The latter are grounded in the common referral of both domains to
social and cultural norms and values. The questions included in the Socratic approach which was applied
in the ethical assessment also explicitly involve18 social and legal aspects. However, overlaps may also
have been influenced by the involvement of the same team member as a researcher in the assessment
of several domains Additionally socio-cultural aspects are part of the context assessment and partially
also presented there. Apart from the limitations described we experienced that group-consultations
should be planned for two hours (especially because of time needed for formalities: agreement,
introduction of interview roles and procedure). A break should be offered. Conducting the consultations in
a team of two researchers (one native speaker and one non-native speaker) ensured mutual
understanding. The consultations were audiotaped twice, which assured the quality of data in the case of
disturbed phone or Skype connections or background noises.
18
When developing the Socratic approach for the ethical assessment, legal and social aspects were not addressed in HTA. With including some of these aspects the Socratic approach aimed to make them more visible in HTA.
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Conclusion
In this part of the case study, we aimed to apply methods developed to assess socio-cultural aspects of
rHBPC (as compared to non-reinforced models of HBPC). rHBPC comprises models of HBPC with an
additional element of caregiver support. This comparison was challenged by the definition of “reinforced
models of HBPC”.
Difficulties resulted from stakeholders’ unfamiliarity with the term ‘reinforced’ models of home based
palliative care. Additionally HBPC was seen as services that generally address the family as a unit and
involve informal carers’ needs, which also blurred the distinction. Nevertheless, some points especially
addressing informal caregivers were brought to our attention as follows.
Relevant from socio-cultural perspective is the social image associated with taking over the role of being
an informal care giver. To be an informal carer can be associated with high social appreciation. A point
that becomes relevant in the political discussions about informal carers in HBPC – (often female) carers
who do care without payment. Another position addresses informal carers as vulnerable group, whose
health and wellbeing has to be protected and explicitly addressed alongside the patients’ needs. This is
especially relevant against the background of socio-demographic challenges which are linked to an
ageing population and the point that in future more very old and non-healthy carers will take over the
caring role. However, medical prescriptions for informal carers (as they could be relevant to access
rHBPC) are viewed critically – e.g. due to related risks of overtreatment and medicalisation.
For informal carers, role conflicts can result from taking over the role of being an informal carer. These
could erase between different roles, e.g. of being a mother, husband and the caring role. rHBPC can help
to support in dealing with role conflicts and maintaining the different roles and protect from social
isolation. As well role conflicts can arise with regard to the caring role only, i.e., when social expectations
(to take over the caring role) conflict with own ideas. The empowerment of informal carers, which is part
of rHBPC, could increase these conflicts and lead to conflicting interests between patients and carers. On
the other hand empowerment can support informal carers to make an informed decision about taking
over or continuing the caring role. Additionally rHBPC aims at supporting informal carers e.g. in
acknowledging and expressing their own needs. This can assist informal carers to fulfil the caring role.
The relationship between patients and informal carers can benefit from the different ways of
empowerment. In addition the way professional carers’ value the role of informal carers and the related
burden can change through rHBPC. However, empowering relatives could also decrease the patient’s
benefit, e.g. if relatives decide to give up the role of being an informal carer.
In general limited knowledge about the options of rHBPC and HBPC can cause social inequalities in
accessing the services.
References
ARELLANO, LE., REZA, M., BLASCO, JA., & ANDRADAS ,E. (2009) A content analysis of health technology assessment programs in Latin America. International Journal of Technology Assessment in Health Ccare, 25(04): 570-576.
DRABORG, E., GYRD-HANSEN ,D., BO POULSEN, P., & HORDER, M. (2005) International comparison of the definition and the practical application of health technology assessment. International Journal of Technology Assessment in Health Care, 21(01): 89-95.
LEE, A,, SKÖTT, LS., & HANSEN, HP. (2009) Organizational and patient-related assessments in HTAs: State of the art. International Journal of Technology Assessment in Health Care, 25(04): 530-536.
LEHOUX, P., TAILLIEZ, S., DENIS, JL., & HIVON, M. (2004) Redefining health technology assessment in Canada: diversification of products and contextualization of findings. International Journal of Technology Assessment in Health Care, 20(03): 325-336.
LYSDAHL, K.B., BRERETON, L., OORTWIJN, W., MOZYGEMBA, K., REFOLO, P., SACCHINI, D., BRÖNNEKE, J.B., VAN DER WILT, G.J., GERHARDUS, A., HOFMANN, B. (2016b) Guidance to assess ethical aspects. In: LYSDAHL, K.B., MOZYGEMBA, K., BURNS, J., CHILCOTT, J.B., BRÖNNEKE, J.B., HOFMANN, B.
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(eds.). Guidance for assessing effectiveness, economic aspects, ethical aspects, socio-cultural aspects and legal aspects in complex technologies [Online]. Available from: http://www.integrate-hta.eu/downloads/
MOZYGEMBA, K., HOFMANN, B., LYSDAHL, KB., et al. (2015) Guidance to assess socio-cultural aspects. In:
Lysdahl, KB, Mozygemba, K, Burns J, Chilcott, JB, Brönneke, JB, Hofmann, B (Eds) (2015) 'Guidance for
assessing effectiveness, economic aspects, ethical aspects, socio-cultural aspects and legal aspects in complex
technologies. [Online]. Available from: http://www.integrate-hta.eu/downloads/
REUZEL, R. (2004) Interactive technology assessment of paediatric cochlear implantation. Poiesis Prax, 2(20):
119-137.
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Legal aspects of (reinforced) home based palliative care
Background
Legal rules have implications for the use of the assessed health technology. For example, legislation can
completely prohibit the use of the health technology, restrict it or regulate it in other ways (for example by
establishing professional requirements for the user). ‘Legal issues’ refers to issues that are regulated by
the law. Legal issues are either of relevance for the use of the assessed technology or they are not
relevant.
Failure to identify legal issues associated with a health technology can result in a violation of the law and
severe legal consequences. Additionally, the law can constitute absolute barriers to the use of a health
technology. Guidance for HTA is important to identify relevant legal issues and allow decision makers to
take appropriate measures to prevent violation of the law.
Aim
To apply guidance developed in the INTEGRATE-HTA project19 that aims to enable non-legal experts to
determine the need for further legal advice or assistance when assessing legal issues relating to
reinforced (rHBPC) and non-reinforced home based palliative care services (HBPC).
Methods
'Guidance for assessing legal aspects in complex technologies' (Broenneke et al. 2015)20 was applied by
two researchers (one English, with experience in palliative care research and one German with
experience in HTA research), experienced in health sciences but with no legal training. Nine legal issues
generally of importance for different technologies (see Box 3.1) were each considered in a 6 step process
as described below. The two researchers applying the legal guidance used their knowledge and
experience of palliative care to inform the responses to questions posed in each of the relevant six steps
of the legal assessment. Users can determine the need for further legal advice or assistance based on
the outcome of the 6 steps.
19
A set of 7 specific questions were addressed in the application process for structuring the feed-back, further details are provided in
'Guidance for assessing effectiveness, economic aspects, ethical aspects, socio-cultural aspects and legal aspects in complex technologies'
(see Lysdahl, KB, Mozygemba, K, Burns J, Chilcott, JB, Brönneke, JB, Hofmann (Eds) (2015) 'Guidance for assessing effectiveness, economic
aspects, ethical aspects, socio-cultural aspects and legal aspects in complex technologies)
20 Brönneke J, Hofmann B, Lysdahl KB et al (2015) Guidance to assess legal aspects. In: Lysdahl, KB, Mozygemba, K, Burns J, Chilcott, JB,
Brönneke, JB, Hofmann, B (Eds) (2015) 'Guidance for assessing effectiveness, economic aspects, ethical aspects, socio-cultural aspects and
legal aspects in complex technologies. [Online]. Available from: http://www.integrate-hta.eu/downloads/
112
Box 3.1 Legal issues of importance for different technologies
Autonomy of the Patient I – Informed Consent
Autonomy of the Patient II – Alternative forms of Consent
Autonomy of the Patient III – Privacy and Data Protection
Market Authorisation I – Medical Devices
Market Authorisation II – Medicinal Products
Clinical Trials
Intellectual Property
Reimbursement in Public Health Care Systems
Special Medical Fields
Steps used in the application of the guidance for each legal issue
1. Users respond to one specific initial question for each legal issue listed in Box 3.1. The response
assesses whether reinforced models of home-based palliative care generally affect the legal
issue.
a) Where the outcome of step 1 indicates that the legal issue is not affected, users continue to
respond to the specific initial question for the next legal issue listed in Box 3.1 until all 9 issues
have been assessed.
b) Where the outcome of step 1 indicates that the legal issue is affected, users continue to
complete steps 2-6 of the guidance for that issue before continuing to assess the next legal
issue listed in Box 3.1.
2. Reading explanations and examples related to the issue being examined - the guidance gives
further explanations of the legal issue and it’s components as well as information on relevant legal
sources/norms.
3. Identifying the most important legal issues affected accordingly to the answer to the initial
question - This information gives users an overview over the importance of the specific legal issue
for reinforced models of home-based palliative care.
4. Reading to identify legal sources (e.g. legal codes) relevant for the affected legal issues
5. Identify relationships with other parts of the HTA
6. Read notice on the when and how to consider the legal issue in an assessment and on the
necessity of professional legal advice.
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Findings
The case study showed that reinforced models of home-based palliative care affect the legal issues of
informed consent (No. 1), Alternative forms of Consent (No. 2), Privacy and Data Protection (No. 3),
Reimbursement (No. 8) as well as the issue of regulation of a Special Medical Field (No. 9).
The first three named aspects are part of the aspect autonomy of the patient which comprises the legal
requirement:
1. to acquire the patient's informed consent,
2. to substitute this informed consent where the patient cannot consent him-/herself, and
3. to protect the patient's data.
Although patient autonomy is generally of relevance for both reinforced and non-reinforced home based
palliative care, all three of the legal issues identified can be potentially more problematic in reinforced
care models. For example, family pressure may oppose the patient's free consent or dissent (concerning
aspect 1); the dual role of family members as carers as well as a source for substituted consent may give
rise to concerns regarding substituted informed consent (concerning aspect 2), and a higher number of
people (family members) may need access to the patient's data (concerning aspect 3).
All legal issues relevant for reinforced care models are also relevant for non-reinforced home based care
models. Whether their relevance is higher for the one or the other intervention cannot be assessed on the
basis of the used guidance. However, there are reasons to assume that the legal issue of autonomy of
the patient is of higher relevance for reinforced home care models. Findings of the case study imply the
need for professional legal advice to achieve legal certainty.
Discussion
We reflected on issues of complexity, integration, stakeholder involvement and the value and application
of the methods developed for effectiveness HTA.
Complexity
Reinforced models of home-based palliative care affect a relatively high number of diverse legal issues.
However, the gravity of these legal issues depend on how specific models are designed and whether
they allow for appropriate answers to legal issues (e.g. whether they include a procedure to ensure
patient’s informed consent). Additionally, the affected legal issues are of varying importance, dependent
on the institutional level on which the HTA is conducted as well as the HTA’s specific question (e.g.
whether conducted for the legislation to clarify reimbursement issues in contrast to being conducted for a
nursing service to clarify procedural questions). A lack of clarity about the technology as well as the
scope of the HTA seem to pose more problems for the assessment of legal issues than issues of
complexity.
Integration
The findings of the legal assessment imply some connections between some legal, ethical and
sociocultural issues, notably with regard to patient autonomy. However the most important finding
regarding the integration of the assessment of legal issues is the requirement to clarify the assessed
technology beforehand. Although patient autonomy is generally of relevance for both reinforced and non-
reinforced home based palliative care, all three legal issues identified in this assessment (i.e. 1) acquiring
the patient's informed consent, 2) substituting this informed consent where the patient cannot consent
him-/herself, and 3) protecting the patient's data) can be potentially more problematic in reinforced care
models.
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For example, family pressure may oppose the patient's free consent or dissent (concerning aspect 1); the
dual role of family members as carers as well as a source for substituted consent may give rise to
concerns regarding substituted informed consent (concerning aspect 2), and a higher number of people
(family members) may need access to the patient's data (concerning aspect 3).
Value and application of methods
The guidance proved useful for identifying legal issues that are or relevance for reinforced models of
home-based palliative care. However, the guidance does not allow any in-depths analysis of the affected
legal issues. However, use of the guidance does not always produce clear and easy to implement results.
The case study showed that legal counsel would have to be sought, as doubts over the specific
consequences of affected legal issues cannot be solved by the guidance.
Conclusions
To avoid problems for the assessment of legal issues, clarification of the details of the assessed complex
technology as well as defining the HTA’s scope (i.e. defining the appropriate policy question) is of
paramount importance for a successful assessment of legal issues.
References
BRÖNNEKE, J., HOFMANN, B., LYSDAHL,B., et al. (2015) Guidance to assess legal aspects. In: Lysdahl, KB,
Mozygemba, K, Burns J, Chilcott, JB, Brönneke, JB, Hofmann, B (Eds) (2015) 'Guidance for assessing
effectiveness, economic aspects, ethical aspects, socio-cultural aspects and legal aspects in complex
technologies. [Online]. Available from: http://www.integrate-hta.eu/downloads/
Moderators of treatment outcome (reinforced home-based palliative care)
Background
Moderators of treatment outcome are factors that influence the effect of a treatment. They can be used to
identify patients that are likely to respond better or worse to a certain treatment. The use of moderators in
research and decision making can result in personalised treatment decisions and optimal care for
individual patients. This can also be applied to the field of palliative care, where patients differ vastly in
backgrounds, social situations, the diseases they have, their needs and problems they face.
Aim
We aimed to find moderators of treatment outcome related to reinforced models of home-based palliative
care using a literature search guidance developed in the INTEGRATE-HTA project.
Methods
To find moderators of treatment outcome in (reinforced) models of home-based palliative care, we
combined a search filter specifically aimed to retrieve moderators of treatment outcome (for more
information see van Hoorn et al., 2015b) with a search strategy to find papers on home-based models of
palliative care based on the search strategy developed by Gomes et al. (Gomes et al, 2013). We
modified Gomes’ search strategy in order to be able to use it with PubMed (the search engine for which
the search filters were created). We also dropped study type restrictions from Gomes’ search strategy to
increase coverage. Identified articles were read through one by one (first on title-abstract only, then full
text) to determine whether they contained information on moderators of treatment outcome concerning
home-based palliative care. A critical appraisal tool (checklist) (for more information see van Hoorn et al,
2015b), was used to critically appraise the selected full text articles.
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Findings
In total 6928 articles were identified using the search methods. A random selection of 3464 abstracts
(50% of the total amount) was taken and were screened on title and abstract, of which 265 were read full
text. Of these full text papers, 67 papers were considered to be relevant. We present here the findings of
34 papers that have been critically appraised (see Appendix 35).
The moderators presented are related to (reinforced) models of home-based palliative care as they do
influence the quality of care for the patient (and partially also the carer). We have identified several
moderators and grouped them according to the outcomes they moderate. Out of the 34 appraised
papers, 12 papers (Li and Chow, 2013, Sinnakirouchenan and Holley, 2011, Cardone et al, 2011, Palmer
et al, 2014, Mills et al, 2009, Wilkinson et al, 2008, Weidle et al, 2006, Rossi Ferrario et al, 2004, Chan et
al, 2008, Hebert et al, 2007, Chow and Wong, 2010, Adamsen et al, 2012) were excluded during the
appraisal process because they did not cover the subject well enough or did not describe or analyse
moderators to such an extent that results were extractable and appraisal was possible.
Summary of identified moderators
See Appendix 35 for an overview of the papers containing moderators for treatment outcome as found in
our review.
Carers’ feelings and psychological health
These are factors that influence the carers‘ feelings of comprehensibility and manageability. That is,
whether carers feel capable and can comprehend the entire situation around the person they are caring
for. Also, factors that influence whether carers have complicated grief after the patient dies.
One interview-based, qualitative study (Milberg and Strang, 2004) among 19 caregivers, identified a
number of factors that had a positive effect on carers’ feeling of manageability: power (i.e. perceived
energy, stamina) of the carer, carers’ competence, accessibility of professionals, support of family,
professionals and others, togetherness (with family and professionals), and open information between
patient, carer and professionals. The study also reported that factors influencing carers’ feelings of
comprehensibility were mixed positive and negative. For example, basic life assumptions, previous
knowledge of the carer, symbols seen by the carer (e.g. signs that signify deteriorations of the patient),
and congruent inner reality (can the carer overview the situation) influence the carers’ feelings of
comprehensibility in both directions, positive and negative.
A German questionnaire-based study (Wiese et al, 2010) among 50 ex-caregivers tried to find factors
that could be used to predict whether carers would suffer from complicated grief after the patient they
cared for died. The relationship between the carer and patient, the satisfaction with the care, the location
of death, psychological stress, duration of care, disease type, and time elapsed since the patient died
were investigated. A significant effect could, however, not be found.
Location of death
Two quantitative studies (one Spanish case-control study under 524 patients (Alonso-Babarro et al,
2013) and one Swedish non-randomised intervention study amongst 280 patients (Ahlner-Elmqvist et al,
2004)) found that patients who live alone or are single are less likely to die at home. A Japanese survey-
based study (Morita et al, 2013) amongst 857 palliative care patients found that home-based palliative
care patients are less likely to die in their own homes when: caregivers are not available; physical
symptoms cannot be controlled; concerns exist about adequate responses to sudden changes of the
patient; or there exists a lack of available physicians. The study did not report effect sizes.
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Survival
Despite the fact that palliative care patients cannot be cured, seven quantitative studies were found that
investigated factors influencing survival (Bengoechea et al, 2010, Cano et al, 2004, Li et al, 2007,
Santarpia et al, 2006, Ruggeri et al, 2013, Pauly et al, 2010, Krishnasamy et al, 2013). Two of these,
(Pauly et al, 2010, Krishnasamy et al, 2013) investigated moderators for death in haemodialysis patients
specifically (in 14,000 Australian patients and 247 Canadian patients respectively). The other studies,
mostly European, were retrospective cohort studies with a number of patients (with advanced cancer,
end-stage respiratory or renal disease and patients with nutritional support) ranging from 13 to 29,348.
Older patients, those with worse physical condition and males were more likely to have a lower survival
probability. Only the physical condition (according to the Karnofski Performance Scale) can be
considered a strong factor (significant relation with survival, though only the study with almost 30,000
patients reports an effect size). The other factors are relatively weak/not always significant (gender) or
dependent on the underlying disease or treatment (age).
Complications
Adverse events or complications are a common reason for treatment failure. They can influence a
patients’ quality of life, general condition, chance for (re)admission to a hospital survival etcetera. The
incidence of complications depends on the specific intervention and patient. Some of these complications
have already been covered by the other outcomes described above (e.g. cardiac death under survival,
hospital (re)admission is likely (partially) covered under location of death as well).
One case review on home parenteral nutrition in 73 patients with amyotrophic lateral sclerosis
(Abdelnour-Mallet et al, 2011) found that if the nutrition catheter is also used for intravenous therapy,
patients have a higher risk on septicemia. They also found a non-significant relation between
frontotemporal dementia and septicemia, but not with other commonly investigated moderators such as
functionality scores or age.
A Nepalese cohort study under 50 patients with home haemodialysis (Sharma et al, 2010) found
significant lower Albumin levels in patients who had peritonitis, but no significant other moderators. An
Italian cohort study under 469 patients with advanced hematological malignancies (Cartoni et al, 2009)
found that the risk on severe thrombocytopenia in patients with acute leukemia is more likely in younger
patients. Lastly, a survey-based study from the United States (Weschules, 2005) on the safety of a
specific nausea-medication combination (8,600 patients) found age to be a moderator for which side
effects occur: those above 65 years of age were more likely to develop nervous-system related side
effects compared to younger patients).
Moderators related to specific interventions
Many palliative care interventions are aimed at improving a specific part of a patients’ burden such as
pain or fatigue. It is therefore logical to assess these interventions only on those specific outcomes.
Ultimately, each of these outcomes will influence the patients’ and their carers’ quality of life. A number of
moderators influence very specific outcomes which are very difficult to merge or generalise with other
outcomes. One observational study under 95 Hong Kong dialysis patients (Zuo et al, 2013) investigated
almost thirty moderators influencing exercise capacity (or more specifically: peak oxygen respiration).
Among these moderators were general moderators such as age, but also very specific cardiac echo
findings such as cardiac dimensions. They found several small effects including age and systolic blood
pressure. Another study amongst 22 chronic obstructive pulmonary disease patients in the United
Kingdom (Chakravorty et al, 2011) investigated several moderators, such as age, gender, and weight, on
the change in sputum volume, but only reported a significant effect of baseline sputum volume.
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A study reporting moderators of costs for haemodialysis based on a Markov simulation (Gonzalez-Perez
et al, 2005) reported that diabetes and age positively influenced cost and a study towards anti-retroviral
treatment at home versus at the clinic amongst 1,453 Ugandan HIV/AIDS patients (Woodd et al, 2014)
fond no effects of disease severity (expressed in CD4 beta-cell counts) on the type and location of death.
Summary of findings from the literature
In general, moderators of treatment outcome can help distinguish which patients benefit most from these
treatments. Moderators may also help explain specific treatment effects found in studies or put them into
perspective. For example, a study may not find a significant effect because the intervention only works in
a specific subgroup of patients. With respect to the question whether or not reinforced models of home
care are more effective than non-reinforced models we saw that most relevant moderators were
associated with the location of death and survival of patients. Only two out of 14 studies discussed
moderators relating to carers: moderators influencing psychological effects such as feeling of
manageability and grief. These moderators are probably most closely related to reinforced models of
palliative care as they directly influence the carer. The rest of our findings are related to specific
interventions, such as haemodialysis and home parenteral nutrition. Unfortunately, none of our
moderators were directly linked to interventions specific to reinforced models of home-based palliative
care.
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ADAMSEN, L., STAGE, M., LAURESEN, J., RORTH, M., & QUIST, M. (2012) Exercise and relaxation intervention for patients with advanced lung cancer: a qualitative feasibility study. Scandinavian Journal of Medical Science Sports, 22: 804-15.
AHLNER-ELMQVIST, M., JORDHOY, MS., JANNERT, M., et al. (2004) Place of death: hospital-based advanced home care versus conventional care. A prospective study in palliative cancer care. Journal of Palliative Medicine, 18: 585-93.
ALONSON-BABARRO, A., ASTRAY-MOCHALES, J., DOMINGUEZ-BERJON, F., et al. (2013)The association between in-patient death, utilization of hospital resources and availability of palliative home care for cancer patients. Journal of Palliative Medicine 27: 68-75.
BENGOECHEA, I., GUTIERREZ, SG., VROTSOU, K., et al. (2010) Opioid use at the end of lifeand survival in a Hospital at Home unit. Journal of Palliative Medicine, 13, 1079-83.
CANO, NJ., PICHARD ,C., ROTH., et al. (2004) C-reactive protein and body mass index predict outcome in end-stage respiratory failure. Chest, 126: 540-6.
CARDONE, KE., MANLEY, HJ., GRABE, DW., et al. (2011) Quantifying home medication regimen changes and quality of life in patients receiving nocturnal home hemodialysis. Haemodialysis International, 1(5): 234-42.
CARTONI, C., NISCOLA, P., BRECCIA ,M., et al. (2009) Hemorrhagic complications in patients with advanced hematological malignancies followed at home: an Italian experience. Leukaemia & Lymphoma, 50: 387-91.
CHAKRAVORTY, I., CHAHAL, K., & AUSTIN, G. (2011) A pilot study of the impact of high-frequency chest wall oscillation in chronic obstructive pulmonary disease patients with mucus hypersecretion. Internal Journal of Chronric Obstructory Pulmononary Disese, 6: 693-9.
CHAN, CT., SHEN, XS., PICTON, P., & FLORAS, J. (2008) Nocturnal home hemodialysis improves baroreflex effectiveness index of end-stage renal disease patients. Journal of Hypertension, 26: 1795-800.
CHOW, SK., & WONG, FK. (2010) Health-related quality of life in patients undergoing peritoneal dialysis: effects of a nurse-led case management programme. Journal of Advanced Nursing, 66: 1780-92.
GONZALEZ-PEREZ, JG., VALE, L., STEARNS, SC., & WORDSWORTH, S. (2005) Hemodialysis for end-
stage renal disease: a cost-effectiveness analysis of treatment-options. International Journal of Technolical
Assessment in Health Care, 21: 32-9.
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KRISHNASAMY, R., BADVE, SV., HAWLEY, CM., et al. (2013) Daily variation in death in patientstreated by long-term dialysis: comparison of in-center hemodialysis to peritoneal and home hemodialysis. American Journal of Kidney Diseases, 61: 96-103.
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119
Patient preferences in (reinforced) home based palliative care (rHBPC)
Background
Patient preferences are increasingly used in medical decisions, e.g. when it concerns the outcomes of a
treatment. These include outcomes such as (risks of) certain adverse events, effectiveness of the
treatment or the overall quality of life a patient has after the treatment. Patients may differ in what
outcomes they regard to be important and for clinical decision making it is important to know what
patients prefer. This can be quite challenging due to the amount of preferences that need to be elicited
(especially in complex interventions such as palliative care), and sometimes the patient simply is not
capable of answering because they are physically incapable or the topic is too value-laden. Therefore, it
may be helpful to see what is already known on patient preferences for treatment outcomes in the
literature: What are common preferences, and where do patients differ in their preferences, and where
not?
Aim
We aimed to identify patient preferences for treatment outcome described in the literature that were
relevant to interventions targeting (reinforced) models of home-based palliative care using guidance
developed in the INTEGRATE-HTA project (Van Hoorn et al., 2015).
Methods
To identify what is known on patient preferences for treatment outcomes, we started with a search
strategy developed by Gomes et al. (Gomes et al., 2013). We translated their search query so it could be
used in PubMed and removed the study type restrictions. To filter out irrelevant papers we combined this
strategy with a search filter on patient preferences for treatment outcome (see guidance on identifying
patient preferences (van Hoorn et al., 2015a)). The set of found papers was subsequently evaluated on
title-abstract, and full text to determine whether the papers contained information on patient preferences
for treatment outcome. Lastly, we applied a critical appraisal tool specifically for studies on patient
preferences for treatment outcomes (for a detailed description of the tool see guidance on identifying
patient preferences (van Hoorn et al., 2015a)).
Findings
By applying the search filters we identified a total of 1606 potentially relevant papers. After evaluating 803
(random selection of 50%) of these on title and abstract, 37 were selected for full text screening, of which
24 contained information on patient preferences for treatment outcomes. Of these, fourteen studies were
critically appraised and the results of the appraisal and data-extraction are presented below. Please see
Appendix 36 for an overview of the 14 appraised papers containing information on patient preferences for
treatment outcome.
There was a mix of qualitative and quantitative research. Both qualitative and quantitative research has
its pro’s and con’s in the context of preference elicitations. Critical appraisal of the selected studies with
design specific criteria showed that overall the studies appeared to be well executed and consistent.
However, overlap in findings of the appraised studies was not high, making it difficult to compare
outcomes between studies. Patient preferences for treatment outcomes were grouped in three main
themes based on overlap found in the reported preferences.
Quality versus quantity of life
We found five studies reporting that most patient prefer quality of life over quantity of life (reporting
percentages ranging from 72-80%) (Miccinesi et al., 2012, McCarthy et al., 2008, Lynn et al., 1997,
Winter, 2013, Barnato et al., 2009). These interview-based studies (four from the United States and one
from Italy) had study populations ranging from 88 to 3357 patients.
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The preference for quality over quantity increases by age and education level (weak relation) and also
when people have a stated belief in life after death (Miccinesi et al., 2012). Significant differences in the
amount of patients who prefer a quality-aimed therapy were found between races and religiosity: blacks,
Hispanics and religious people are more likely to prefer therapies aiming for longer life, according to three
interview-based studies from the United States (Barnato et al., 2009, Winter, 2013, Volandes et al., 2011)
under 2847, 87, and 304 persons. However, with the exception of four studies (Lynn et al., 1997,
Strachan et al., 2009, Miccinesi et al., 2012, Foo et al., 2012), all studies we found covering quality
versus quantity of life preferences did not use patients in a palliative care phase (some only interviewed
elderly regardless of having any disease). Instead, these studies provided hypothetical situations or
choices to their participants and investigated the preferences. As these participants may think differently
when they are themselves in a palliative care phase, one should be careful to extrapolate such findings to
the palliative care setting.
Location of death or treatment
We found evidence in four survey-based studies (2847 elderly people from the United States (Barnato et
al., 2009), 106 Canadian patients with advanced heart failure (Strachan et al., 2009), 371 South Korean
cancer patients (Choi et al., 2005), and 258 Japanese cancer patients (Ishikawa et al., 2013)) that
patients most often prefer to die at the comforts of their own homes over hospitals or hospices. However,
patients do differ in their views on the importance of where they ultimately die. Blacks and Hispanics were
found to have a less strong preference for death at home compared to whites (versus in the hospital or
hospice) (Barnato et al., 2009). The Canadian study (Strachan et al., 2009) found that one quarter of
patients had no preferences on where they would ultimately die and that location of death is not the most
important outcome of home-based palliative care. The Japanese survey-based study (Ishikawa et al.,
2013) found that about half of the patients and their families preferred to die at home. This finding was
supported by the Korean survey-based study (Choi et al., 2005). They found the most important
argument for home-death preference to be able to live out their lives in privacy and to be with their family
when their life ended. For those who preferred to die at the hospital instead, the availability of medical
treatments and reduction of burden for carers were the main arguments. One study found that receiving
palliative chemotherapy influencing dying at the preferred location of death (Wright et al., 2014), but
preference data could not be obtained from the study.
Treatment specific preferences
This theme is a collection of preferences for specific outcomes relating to specific interventions. For
example, treatment aims for 106 Canadian home-based palliative care patients with advanced heart
failure were found to be: reduction of emotional, physical burden on family; reduction of symptoms; and
especially adequate communications (Strachan et al., 2009). These factors are much more often
considered important by heart failure patients than: having the same nurses looking after the patients
(continuity of care); to have spiritual and religious needs met; and to die in the location of their choice.
Similar findings were found by a study under 42 veteran hospice patients (Kumar et al., 2011). Another
study reported on preferences for receiving chemotherapy, describing that patients preferred avoidance
of toxicity of the chemotherapy over receiving the treatment at a specific location (home or hospital)
(Kumar et al., 2011).
Summary of findings from the literature
Preferences for treatment outcome are important to determine the value of a treatment. Interventions are
likely to cause multiple outcomes, some considered good (e.g. pain control), other bad (e.g. adverse
effects). If an intervention excels at pain control but comes with more adverse effects, the preferences
between these outcomes determine the overall value of that treatment. Furthermore, if there is
heterogeneity in the way patients value these outcomes, eliciting these preferences becomes important
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for clinical decision making. Identifying patient preferences for treatment outcomes is also important in
determining the acceptability of treatments.
Treatments should be evaluated on treatment outcomes deemed important to the patient. This also
applies to which treatment outcomes are investigated in studies. We identified three groups of
preferences concerning home-based palliative care: quality of life versus quantity (length) of life, location
of death and (treatment-specific) care goals.
We found evidence that most patients prefer treatments focusing on quality of life over quantity of life, but
cultural differences do exist. The same applies to preferences on where a patient dies: most patients
prefer not to die at the hospital. In some papers patients reported that other factors of the treatment, such
as the reduction of emotional or physical burden on family and symptom relief), were of higher
importance than where the patient ultimately died. These differences prompt individual assessment on a
patients’ preferences, by active elicitation if possible.
SAP Involvement
A summary of our results was presented to an advisory panel to relate the findings to the daily practice:
that is, to determine whether the findings made sense and were not missing important issues, whether
the findings were usable and relevant. The advisory panel consisted of two ex-carers and five
experienced palliative care nurses. (See Appendix 37 for further details about the stakeholders involved
in validating the patient preferences and moderators of treatment effect).
When the findings on moderators of treatment outcome were presented to the advisory panel, we found
recognition in most of these findings. However, the advisory panel did point out that as every patient and
setting is different, individualised care is extremely important. To do so, it is important to know the
individual patients’ needs and preferences in order to determine what the best treatment is for that
patient.
After presenting the results concerning moderators on carers’ feelings of manageability and
comprehensibility, one carer noted that support of the environment (friends, family, medical professionals,
etc) was in his view of especial importance (e.g. supporting day-to-day chores). It was the advisory
panel’s experience that the important aspects in home-based palliative care are not so much the factors
underlying medical decisions, but more so the organisation around the palliative care patient in general. It
was pointed out that experience of the carers and professionals and their communication skills are very
important to not only organise the care around the patient, but also to support the patient better
(understand what (s)he needs). The nurses explained that good nursing is about relationships, listening,
and communicating. Cultural differences, as found in our evidence, were recognised to be important
factors in patient preferences: not only as stratifying patient preferences, but also the effect cultural
differences have on the communication (e.g. able to sense and discuss issues and whether the patient or
carer wants to talk about it). For instance, one nurse explained that death is recognised as a failure in
some cultures. This makes it difficult to discuss end-of-life issues and care focusing on quality rather than
length of life with these people.
When we presented the findings on patient preferences for treatment outcome, one nurse was interested
in the fact that the majority of patients preferred a treatment aimed at quality of life over quantity of life.
The nurses described that they see a lot of patients receiving care aimed at life lengthening (e.g.
chemotherapy), which the patients regretted in hindsight. The preferences for location of death were
recognised by one of the carers as important, stating that it is important to avoid the hospital, for instance
at the emergency department of a hospital, as a location where a patient eventually dies.
In summary, the advisory panel recognises and supports the findings we presented them, but at the
same time regard additional factors to be important when it comes to providing good home-based
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palliative care: experience of the professionals and carers, communication and networking skills. Though
nursing is built upon scientific evidence, other factors exist that influence the quality of care.
The discussions in the advisory panel showed that even though we can determine what on average the
preferences of patients are, there will always remain differences between patients and it remains
important to assess the preferences of individual patients.
Reflections on the application of the guidance to assess moderators of treatment outcome
and patient preferences
We reflected on issues of complexity, integration, stakeholder involvement and the value and application
of the methods developed for effectiveness HTA.
Complexity
Home-based palliative care is a good example of a complex intervention. This is shown by the diversity of
moderators and preferences we found. For instance, the moderators we found relate to interventions
such as parenteral nutrition, haemodialysis, and anti-retroviral treatment. All these interventions may fall
under home-based palliative care, but it depends on the exact patient group as to whether the specific
moderators are relevant or not. The same applies for patient preferences. Though they showed some
similarities, it is still rather difficult to extrapolate these preferences to palliative care in general.
Furthermore, the advisory panel discussions showed that non-scientific factors, such as experience and
communication skills, often play a role in applying evidence in practice. These issues add up to the
complexity of home-based palliative care: (1) the diversity of evidence makes it difficult to aggregate
evidence that can be applied to the whole target group; and (2) the need to determine applicability of
evidence on a case-by-case basis.
Integration
Findings about patient preferences and moderators of treatment effect suggest there are links with other
research areas. For instance, cultural differences in end-of-life decisions prompt the integration of socio-
cultural properties with patient preferences. It should also be obvious that integration with evaluations of
costs and effects is needed in order to identify valuable technologies in view of patient preferences and
moderators.
Stakeholder involvement
Stakeholders verified the information presented and provided additional insights into home-based
palliative care. (See Appendix 37 for a summary of stakeholder feedback about their involvement in the
meeting to discuss the analysis).
Value and application of methods
Patients in (home-based) palliative care are characterised by a large heterogeneity. This heterogeneity
influences treatment outcomes and their valuations. Acknowledging these effects, the use of technology
can be optimised by targeting technologies to those subgroups where they work the best. Thus, insight in
moderators and patient preferences for treatment outcomes will help to evaluate as well as implement
personalised healthcare.
Preferences and moderators for treatment outcomes are found in studies with a diverse set of study
designs. This has made us decide to perform broad searches for moderators and preferences relating to
models of home-based palliative care, not specifically reinforced models. The result is that we found a
large set of papers with different moderators and patient preferences. Their diversity made it difficult to
relate these with each other for the comparison of reinforced and non-reinforced home-based palliative
care. Moderators for treatment outcomes are treatment and disease specific, which makes it difficult to
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translate moderators to other treatments or settings. Preferences for treatment outcomes are easier to
translate to different settings as they are more generic. However, cultural differences and other causes of
heterogeneity herein need to be taken into account as well.
Conclusions
We were able to identify several moderators and patient preferences for treatment outcome related to
(reinforced) models of palliative care. The advisory panel supported these findings but identified
additional factors that were important in their view. This shows that it is important to bring together the
findings from literature and daily practice. Doing so can provide the most optimal care for home-based
palliative care patients and improve personalised healthcare.
References
BARNATO, AE., ANTHONY, DL., SKINNER, J., et al. (2009) Racial and ethnic differences in preferences for end-of-life treatment. Journal of General Internal Medicine, 24: 695-701.
CHOI, KS., CHAE, YM., LEE, CG., et al. (2005). Factors influencing preferences for place of terminal care and of death among cancer patients and their families in Korea. Support Care in Cancer, 13: 565-72.
FOO, AS., LEE, TW., & SOH, CR. (2012) Discrepancies in end-of-life decisions between elderly patients and their named surrogates. Annals of Academic Medicine Singapore, 41: 141-53.
KUMAR, G., MARKERT, RJ., & PATEL, R. (2011) Assessment of hospice patients' goals of care at the end of life. American Journal of Hospice and Palliative Care, 28: 31-4.
LYNN, J.,TENO, JM., PHILLIPS, RS. (1997) Perceptions by family members of the dying experience of older and seriously ill patients. SUPPORT Investigators. Study to Understand Prognoses and Preferences for Outcomes and Risks of Treatments. Annals of Internal Medicine, 126: 97-106.
MCCARTHY, EP., PENCINA, MJ., KELLY-HAYES, M. et al (2008) Advance care planning and health care preferences of community-dwelling elders: the Framingham Heart Study. Journals of Gerontology Series: Biological Sciences and Medical Sciences, 63: 951-9.
MICCINESI, G., BIANCHI, E., BRUNELLI, C., & BORREANI, C. (2012) End-of-life preferences in advanced cancer patients willing to discuss issues surrounding their terminal condition. European Journal of Cancer Care (England), 21: 623-33.
STRACHAN, PH., ROSS, H., ROCKER, GM., et al. (2009) Mind the gap: Opportunities for improving end-of-life care for patients with advanced heart failure. Canadian Journal of Cardiology, 25: 635-40.
VAN HOORN, R., KIEVIT, W., BOOTH, A., et al. (2015) The retrieval and critical appraisal of literature on patient preferences for treatment outcomes [Online]. Available from: http://www.integrate-hta.eu/downloads/ [Accessed: 28
th September 2015].
VOLANDES, AE., FERGUSON, LA., DAVIS, AD., et al. (2011) Assessing end-of-life preferences for advanced dementia in rural patients using an educational video: a randomized controlled trial. Journal of Palliative Medicine, 14: 169-77.
WINTER, L (2013) Patient values and preferences for end-of-life treatments: are values better predictors than a living will? Journal of Palliative Medicine, 16: 362-8.
WRIGHT, AA., ZHANG, B., KEATING, NL (2014) Associations between palliative chemotherapy and adult cancer patients' end of life care and place of death: prospective cohort study. British Medical Journal 348: g1219.
124
Context and implementation for complex interventions
A qualitative review - contextual enablers and barriers to implementation
Background
Interactions between the intervention, implementation and context may be effect-modifying (Rychetnik et
al., 2002). Context and setting refer to the place and time from which the evidence for the HTA report has
come and/or in which the HTA report will be used (EUnetHTA, 2007). Setting in particular is commonly
used in HTA to refer narrowly to an organisational dimension of health care, such as primary, secondary
and tertiary care, or community care (EUnetHTA, 2007). Context is often used synonymously with setting
and environment (Kitson et al., 1998, McCormack et al., 2002), embracing static (e.g. physical
environment) and dynamic aspects (e.g. relationships, networks) as well as the theory underpinning the
intervention and its implementation (Pawson et al., 2005). Context is defined as a set of characteristics
and circumstances that consist of active and unique factors that surround the implementation effort
(Pfadenhauer et al., 2015). As such it is not a backdrop for implementation but interacts, influences,
modifies and facilitates or constrains the intervention and the implementation effort (Pfadenhauer et al.,
2015). As such context embraces the setting of care as well as roles, interactions and relationships
between different factors within the context (Pfadenhauer et al., 2015)). Implementation is defined as the
process, constellation of processes or means of assimilating or putting into use an intervention - either
evidence-based or theory-based – in an organisation or a setting (Damschroder et al., 2009,Rabin et al.,
2008, Klein & Sorra, 1996). Implementation can be considered as an actively planned and deliberately
initiated effort with the intention to bring a given object into practice (Pfadenhauer et al., 2015). These
efforts are undertaken by agents, who are either actively promoting the use of the intervention or who
adopt the newly appraised practices ((Pfadenhauer et al., 2015). Implementers are challenged by two
conflicting demands: on the one hand, universal interventions are to be implemented with fidelity, on the
other hand, these must be adapted to local needs and circumstances. Therefore, it is critical that the
intervention, its context and implementation are documented transparently in both primary studies and
systematic reviews (Wells et al., 2012).
For a variety of reasons, interventions aimed at improving palliative care services encounter challenges
beyond the usual problems of implementation of complex interventions (van Riet Papp et al., 2014).
Palliative care interventions focus on patients with changing and increasing needs for treatment and
support, suffering from multiple symptoms and receiving care from a wide range of professionals (van
Riet Paap et al, 2014; Vissers et al., 2013). This requires a high degree of collaboration between all
stakeholders involved (Radbruch & Payne, 2009). Complex interventions are known to interact with the
context in which they are implemented (Cambon et al., 2012), and both context and implementation act
as modifier to the effectiveness of complex interventions (Rychetnik et al., 2002). One obstacle to the
reporting and documentation of barriers and facilitators to the implementation of complex interventions is
the difficulty of identifying and quantifying factors that might be relevant for the effectiveness of an
intervention (Wells et al., 2012). Qualitative research can fill these gaps by providing valuable insights
enabling the replication and transfer of evidence-based programmes by identifying barriers and
facilitators to the implementation of complex interventions (van Riet Paapet al., 2014), forming the first
step in systematically overcoming these challenges (Grol, Wensing & Eccles, 2005).
Aim
The aim of this review was to identify enablers and barriers of context and implementation of home-based
palliative care services in Europe using an unpublished framework assessing the context and
implementation of complex interventions (CICI) developed in the INTEGRATE-HAT project (Pfadenhauer
et al., 2015) (see Appendix 38 for more information about the CICI framework).
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Methods
We conducted a systematic review any type of primary study that uses qualitative methods for both data
collection and data analysis or mixed methods studies that allowed the extraction of primary qualitative
findings. Appendix 39 provides a detailed account of the methods used. Four data bases were searched;
MEDLINE, EMBASE, PsycINFO and CINAHL for studies focusing on models of palliative care for adults
(>18 years) where they reported the perceived and experienced barriers and facilitator of home-based
palliative care. We paid particular attention to the subgroup of family caregivers as embraced in
‘reinforced’ home-based palliative care models. Participants in primary qualitative research included
patients, informal caregivers, professional caregivers, or any other stakeholder involved, targeted or
affected by a home-based palliative care intervention. We included studies published until January 2015.
One reviewer assessed titles and abstracts of the 5,531 records found to evaluate their potential
eligibility. The full text of potentially eligible records was independently screened by a second reviewer.
Subsequent data extraction was performed by one author using a data extraction form based on the CICI
framework (Pfadenhauer LM et al. unpublished framework on Context and Implementation for Complex
Interventions (CICI)).
We applied “best fit” framework synthesis (Carroll C et al., 201; Booth, 2012; Carroll, Booth & Cooper ,
2011) as the method for analysis using the CICI framework as an a priori framework for synthesis.
The quality of the included studies was assessed using a quality assessment process as suggested by
Carroll et al. (2014) (Carroll, Booth & Lloyd-Jones, 2012). The tool comprises four questions assessing
the adequacy of reporting of methods and procedural elements, which represent items from the Critical
Appraisal Skills Programme (CASP) tool as well as the Evaluation Tool for Qualitative Studies (see Table
A39.1 in appendix 39).
Results
In total, 5,531 studies were identified from which 2,543 duplicates were removed. From the 2,988 records
screened, 2,792 were excluded, and data from 41 full-text articles that met the inclusion criteria were
extracted (see Figure A39.2 in appendix 39). 19 studies were conducted in the UK, 11 in Sweden, three
in the Netherlands, Denmark and Belgium, one in Italy and one including participants from the
Netherlands, Belgium and the UK. In 21 studies, patients received regular HBPC while in 20 studies,
families were the unit of care that received rHBPC. In 9 studies, participants reflected on the
phenomenon of HBPC in general. In 19 cases, the study was conducted while the patient was still alive
while in eight studies, the patient had deceased already. 15 studies showed only the perspectives of
professional caregiver, 11 showed the perspectives of informal caregivers, three the perspectives of
patients and 12 explored multiple perspectives. The characteristics of included studies are shown in
Table A39.3 in Appendix 39)
Quality of reporting
Seven studies were inadequately reported ( Harding et al., 2012; Bertero, 2011; Carlsson, 2014; Milbert
& Strang, 2000; Pype et al., 2013; Slort et al., 2011; Wilson et al., 2014), while 34 were adequately
reported. For more details, please view TableA39.4 in Appendix 39).
Summary of findings
Figures 3.8 and 3.9 respectively show the categories that emerged for the single domains of the context
and implementation component of the CICI framework. A description of each domain is provided in the
CICI framework guidance (Pfadenhauer et al., 2015) and Appendix 39. The epidemiological domain was
not served by any included studies and therefore does not appear in the text about context.
126
Access to health care is affected by a number of contextual factors (e.g. geographical, socio-economic,
and political) and is therefore presented in links several domains of context. Appendix 39 presents more
detailed findings with accompanying data to illustrate each domain and the respective categories and
themes.
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Context
All categories and themes that were assigned to context domains are shown in 3.8
Figure 3.8 Domains, Categories and themes of the context dimension of the CICI framework
Locational domain Geographical domain
Epidemiological domain
Socio-cultural domain
Hospitalization of home
Effect of setting
Modification of social role at home
Access to health care system
Infrastructure
Family context
Socio-cultural environment
Socio-economic domain
Socio-economic status of recipients of care
Economic climate
Access to health care system
Legal domain
Decision-making in sedation
Sharing of information
Political domain
Access to health care system
Ethical domain
Autonomy
Privacy
Moral stress
Conflicting interests
Social understanding of health issue
Ability to care
Assumption of Roles
Decision making
Social support
Social control and expectations
129
Location
Being at home had a relieving effect on the patient, giving him a sense of security and community. Home
implied some kind of normalcy that was appreciated by both the patient and the lay caregiver.
“…It’s easier to be unwell if I can stay at home…”
(Patient, Sweden, rHBPC) (Appelin & Bertero, 2004)
When home is transformed into a “hospice at home”, the location requires both the patient and informal
caregiver to adopt specific roles.
Geographical
Issues were reported in reference to access to health and social services. Access to health care system
was a recurrent theme that according to its multilevel character was attributed to more than one domain,
i.e. the political and socio-economic domain. For example, in Italy, strong differences between the South
and the North were observed.
“We need a standard level of services in all districts; [services] shouldn’t change depending on where you happen to live”
(Caregiver, Italy) (Borreani et al., 2014).
An additional barrier to HBPC was imposed by long distances and lack of infrastructure to deal with this
issue.
Socio-economic
The socio-economic status of recipients influenced the delivery of care, with professionals highlighting
difficulties created by social deprivation.
“It’s very difficult when there are people surrounding who are inebriated, and there’s quite a
lot of violence; they’ve got a big dog in the kitchen, barking, and drunken people shouting
abuse at each other when I was trying to sort out this lady who had cancer”
(Professional caregiver, United Kingdom, HBPC) (Wilson et al., 2014)
Many authors reported an economic climate of financial constraints whereas different mechanisms were
installed to facilitate HBPC (e.g. early support visits in the U.K.) and get people into homecare (e.g.
reduction of hospital beds in Sweden).
Socio-cultural
Family have a unique role in palliative care, being both a provider and a recipient of palliative care.
Accepting the terminal diagnosis of a loved one and developing the ability to provide care at home were
preconditions for providing HBPC. Families that were able to cope well with the new situation have
specific traits and characteristics, such as stability of family relationships and the confidence to ask for
help. The change in lifestyle required patients and informal caregivers to take in new roles and decision
making became an issue for the whole family. Surrounding the micro context of the family, the community
also provided support. However, the community in which the family was embedded also exerted influence
on the decisions of the families and members thereof to provide HBPC:
“Ties between us are very strong. Too strong. […] Social pressure from amongst Turkish
people also plays a role.”
(Netherlands, Lay caregiver, rHBPC) (De Graaff & Francke, 2003)
130
Political
Access to the health care system was affected by the accessibility of the health care system and the
utilisation of services that was which was particularly difficult for some families (e.g. non-native speakers
of the country) and hindered by bureaucracy.
“You have to wait for ages on the phone, and then they can’t give you an appointment for
months or years… I used to get a check-up every six months, and have a blood test more
easily. [Now there’s] too much bureaucracy”!
(Patient, Italy, rHBPC) (Borreani et al., 2014)
Legal
Issues evolved for professional caregivers especially in regard to decision making about sedation.”
Additionally, respecting a patient’s rights while also considering relatives’ wishes was a legal challenge
for professional caregivers.
“I said 'I will follow the wishes of your husband and we will start the procedure to eventually, um, let it happen'... so she wasn't very happy with that. But in fact, her husband was the
patient, not her. So of course I followed the viewpoint of that man."
(Professional caregiver (GP), Belgium, rHBPC) (Pype et al., 2013)
These legal issues affected the ethical and socio-cultural domains of context.
Ethical
Issues related to privacy for all members of the household in which palliative care is provided. The
intrusion of the family’s home also imposes an additional burden on the professional caregiver. The
autonomy of the patient can be compromised by relatives who want to withhold information in order to
protect the patient. Withholding information can delay the patient accepting the diagnosis and interfere
with the planning of future care.
‘‘I had a case where she was protecting him and he wasn’t accepting’’
(Professional caregiver, United Kingdom, HBPC) (Wilson et al., 2014)
A conflict of interest may mean moral stress is experienced by all stakeholders (i.e. patients, informal
caregivers and professionals). Professional caregivers are torn between interests of the patient and his
family. The informal caregiver does not want to appear selfish and therefore withholds his needs whilst
the patient does not want to be a burden on his family, the professional caregiver or society. Professional
caregivers experience conflicting interests, in some situations not knowing who to take care of first.
Epidemiological
We did not identify any aspects regarding the epidemiological context.
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Figure 3.9 Domains, Categories and themes of the implementation dimension of the CICI framework
Implementation
Policy
Meeting a society’s demands Facilitation of death at home
Navigating the health care system
Social protection of informal caregiver
Funding
Financial support of informal caregivers
Availability of resources for care provision
Not being worth the investment
Provider
Characteristics of provider
Accessibility of professional caregiver
Attitude, behaviour and personality of professional
caregiver
Confidence and security
Personal networks
Knowledge and competence
Team dynamics
Tasks, time pressure and workload
In-team communication
Interaction with care recipients
Communication with care recipients
Relationships
Interpersonal continuity
Training needs
Training needs in terms of care provision
Training needs in terms of lay caregiver support
Organisation & Structure
Supervision and guidance Continuity
Organizational continuity
Coordination
Collaboration and cooperation
Continuity of staff
Informational continuity
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Implementation
All domains with respective categories and themes identified with respect to the implementation
dimension are shown in Figure 3.9.
Policy
Several studies reported that policies were installed to meet a society’s demand for HBPC and to
facilitate death at home where this was preferred by patients. However, more policies were needed for
informal caregivers as highlighted in an Italian study (Borreani et al., 2014). Informal caregivers frequently
reported that they needed support in navigating their respective health care system and that information
should be made accessible more easily. Facilitators in navigating the system were knowledge of where to
seek information, making use of available services, confidence to ask for help, and being proactive in
seeking information (Ryan et al., 2008).
Funding
Funding was mainly criticised by study participants (Carroll, Booth & Cooper, 2011; Anquinet et al., 2015;
Ryan et al., 2008). In the UK and Italy, informal caregivers did not feel sufficiently supported by the health
and social system and reported shortages as well as perceived refusal of reimbursement. Professionals
partly felt anxious and disappointed by their organizations (Harding et al., 2012; Borreani et al., 2014)
when they reported lack of personnel and time (Bertero, 2010) resources that they had to compensate for
(Grande et al., 2004).
“I feel that I do not have the time to give the kind of care that I would like to give. . . . I do
not have the possibility of giving that kind of care. As I experience it, the biggest problem
is that we do not have the personnel; in fact, we do not have any more resources. We
have what we have”.
(Professional caregiver, Sweden) (Bertero, 2002)
Moreover, patients reported they felt not being worth the investment (Ryan et al., 2008) if, for example,
promised items such as a wheelchair were not provided, or if they perceived that reimbursements or
equipment were being held back from them (Borreani et al., 2014; Jarrett, Payne & Wiles, 1999).
Organisation and structure
Undermined by lack of infrastructure and immediate support (35), care was described as ‘disjointed’ by
professionals (Dunne, Sullivan & Kernohan, 2005):
“If there was one person that would be responsible for the overall care of the patient, then
communication might be better. It would be much better if there was one designated person
to take responsibility for the patient’s care. In that way you would know who was involved
and exactly when they were involved. At the moment it’s very disjointed”.
(United Kingdom, Professional caregiver, HBPC) (Dunne, Sullivan & Kernohan, 2005)
Alongside coordination of care (Dunne, Sullivan & Kernohan, 2004; Neergaard et al, 2010; Seamark et
al., 2014; Walshe et al., 2008), that required assigned responsibilities (Dunne, Sullivan & Kernohan,
2005; Neergaard et al., 2010;Walshe et al, 2008) and defined processes (Harding et al., 2012; Jarrett,
Payne & Wiles, 2999;Goldschmidt et al., 2006; Grande et al., 2004; Jack et al., 2013; Seamark et al.,
2014), collaboration and cooperation between stakeholders (Grande et al, 2004) were important themes.
Continuity was the most prominent theme (Milbert & Strang, 2000; Appelin, Broback & Bertero, 2005;
Jarrett, Payne & Wiles, 2999; Dunne, Sullivan & Kernohan, 2005; Devlin & Mcllfatrick, 2010; Seamark et
al., 2014; De Bock et al, 2011), and it’s importance was recognised, especially with regard to the
organization, staff and information.
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Organisational continuity benefitted from coordination with assigned responsibility to individuals (Dunne,
Sullivan & Kernohan, 2005), providing access to professionals (Bertero, 2011; Weibull, Olesen &
Neergaard, 2008; Neergaard et al, 2010; Seamark et al., 2014). Staff continuity was an important
facilitator in networking with professionals to provide home-based palliative care (Ryan et al., 2008).
However, bureaucracy was identified as one barrier to continuity of staff (De Graaff & Francke, 2003)
whilst lack of staff continuity undermined communication (Jarrett, Payne & Wiles, 2999; Dunne, Sullivan
& Kernohan, 2005). Lack of informational continuity was experienced by some professionals, informal
caregivers and patients. Informal caregivers often had to transmit information between different providers
of HBPC, although some informal caregivers were impressed by the continuity of information.
Mechanisms facilitating informational continuity included regular meetings, updates and the use of
modern technology. Institutionalised access to guidance, advice and supervision also enabled HBPC to
work. Supervision and guidance should be part of the infrastructure as these were relevant factors to the
implementation of (reinforced) HBPC. Supervision and guidance should be made available to both
formal (i.e. professionals) and informal caregivers at all times to increase feelings of security:
“I believe the palliative team can do things that I can’t . . . I’ve had contact with them several
times concerning pain therapy. It’s made me feel more secure”.
(Professional caregiver, Denmark, HBPC) (Neergaard et al., 2010)
Provider
Palliative care encompasses not only technical and medical services but also needs to cater to recipients’
psychological, social and spiritual needs. Therefore, the relationship between the professional caregivers
and the lay caregiver and patient must be based on mutual acceptance and trust. Transforming a
relationship “from stranger to trusted helper” (Choi et al, 2005 takes time (Bertero, 2011) and continuity
(Saultz, 2003) as well as a professional caregiver who is sensitive (Ingleton et al., 2011), attentive
(Griffiths, Ewing & Rogers, 2013) and can adapt to the different family contexts and needs. The
characteristics of the individuals delivering palliative care, including their behavior knowledge, skills, and
training are important influences on relationships. Professionals who were flexible and adapted to the
patient’s changing conditions and needs and physicians who offered their help without having to be
asked for it were also valued (Jarrett, Payne & Wiles, 1999). Professionals who were extremely
dedicated to their work exceeded recipients’ expectations and were highly praised (Grande et al., 2004;
Exley & Tyrer, 2005). Additionally, accessibility was an important issue for professionals and informal
caregivers (Grande et al., 2004), providing a sense of security to informal caregivers. Informal caregivers
valued professionals who engaged in joint problem solving (Jarrett, Payne & Wiles, 2999; Grande et al.,
2004; Ingleton et al., 2011), had a caring attitude, were supportive, respectful, kind and sympathetic
(Grande et al., 2004).
"I mean [you’re] totally yourself with them... even when - the day she was dying when they
came, they were still very respectful, […] They were just out of this world... they were
fantastic.”
(Lay caregiver, UK, rHBPC) (Ingleton et al., 2011)
Joint problem solving and mutual support enhanced team working (Pype et al., 2013) and learning
opportunities for professionals (Pype et al., 2014). However, conflicts and tensions within multidisciplinary
teams resulted from an unclear division of competencies (Pype et al., 2013), lack of confidence in others’
competence (Pyape et al., 2013; Holmdahl, Savenstedt & Imoni, 2014), and exclusion from the decision
making process. Professionals who were considered to be disrespectful to patients and colleagues were
a source of stress (Neegaard et al., 2010). Informal caregivers’ confidence in the quality of care was
compromised if they had cause to question the professionals’ knowledge, skills or competence
(Goldschmidt et al., 2006; Seamark et al., 2014).
134
Professionals also reported difficulty in establishing relationships with clients (Griffiths et al., 2007),
occasionally due to the patient’s attitude (Borreani et al., 2014).
Interactions between domains
As expected, there were overlaps between single domains as well as categories that were attributed to
several domains (e.g. access to health care). The links between the specific domains were flagged
whenever an excerpt was assigned more than one code.
“Access to health care system” as a category emerged as a link between several domains of context. On
the one hand, access to the health care system can be enforced by a political system and facilitated or
hindered by geographical issues. On the other hand, the utilization of the health care system is influenced
by organizational (e.g. continuity of care), socio-cultural (e.g. language barriers, attitude towards health
care system, equality) and socio-economic context (e.g. advancing of equipment) as well as provider-
based (e.g. commitment of professional caregiver to support navigation through health care system)
factors. Accessibility and utilization are also influenced by policy (navigating the health care system),
socio-cultural aspects (e.g. language barriers, social inequality), as well as ethical aspects (e.g. equality,
justice and moral stress).
The locational domain relates in particular to the socio-cultural (family context) and ethical domain
(privacy, moral stress).
The socio-cultural domain was highly interactive with the ethical domain, in particular in terms of family
context with the categories of moral stress and privacy. For example, the perceived obligation to care
was often influenced by inner-family roles as well as social control and expectations. Moreover, gender
was related to taking in a lay caregiver role which again affects the autonomy of informal caregivers.
In addition to the geographical domain, the socio-economic domain interacted with the socio-cultural,
provider and funding domain of the CICI framework.
The political domain interrelates with the organisation & structure domain, especially in terms of
facilitating or hindering continuity of care in terms of providing a functional health care system. In terms of
resource allocation, the political domain was also related to the ethical domain where this allocation could
cause moral distress in the professional caregivers.
Apart from the interaction with the socio-cultural domain, the ethical domain showed strong links to the
provider domain. Conflicting interests were experienced by professional caregivers and caused moral
stress. The intrusion into privacy linked to both the provider and the locational domain. Moral stress was
also related to the organisation as well as policy, from where interests were imposed on the professional
caregiver. Additionally, funding restricted the abilities and especially time the professional caregiver had
available when providing care, which in return caused moral stress.
In the legal domain, links to the ethical domain became apparent, especially with regards to moral stress
and conflicting interests for the professional caregiver. Team dynamics (provider domain) sometimes
compromised by legally binding decision making in sedation.
The provider domain interacted with all domains due to being the delivery agent of palliative care. As
such, the provider domain was affected by all factors present in the context domains. Of particular
relevance were the organization and structure, the ethical and socio-cultural domains.
Organization and structure exerted a particular influence on the provider. By not providing clear
processes and responsibilities, a lack of coordination could cause moral stress as well as conflicting
interests in the professional caregiver. Naturally, the continuity of care was influenced by the overall
health care system and the political domain.
135
Policies could be installed in order to get organisations to provide better structures and mechanisms to
allow continuity of care and sufficient funding could provide the resources to guarantee continuous care
provision. Additionally, by integrating the lay caregiver and relatives into care provision, the organisation
domain also interacted with the socio-cultural domain.
Funding and policy were highly interactive, mostly because funding was dependent on policies and policy
directives. Both domains influenced the organisation and the provider, thus affecting the socio-cultural as
well as ethical domains.
Discussion
To our knowledge, this is the first qualitative review assessing barriers in context and implementation that
were of relevance for rHBPC. We revealed a variety of factors in the context that negatively or positively
affected the implementation of a respective intervention.
Home based palliative care – with or without a reinforced element - is a peculiar example for a complex
intervention as the intervention, context and implementation are mutually intertwined, creating complex
pathways for mechanisms of action. Not only are the interventions very heterogeneous in terms of their
design and delivery, so are the care settings.
A particular insight was the importance of the micro context surrounding the patient and their family.
Therefore, one of the most populated domains of context was the socio-cultural domain, which family
context was attributed to. In this micro context, most of the interaction between patients, informal
caregivers as well as professional caregivers took place. This interaction was a particular predictor for the
success of a HBPC intervention, emphasising also the importance of the domain of the provider of HBPC
– the professional caregivers.
Some domains of context were not served by primary studies. For example, reflections on the political,
socio-economic as well as policy domain were mainly derived from the interpretations of primary study
authors while the socio-cultural, ethical and the provider domains were supported by both verbatim
quotations and authors’ interpretations.
While the evidence basis for some categories was very rich (especially for the provider domain), some
domains were hardly served by the data. Thus, the geographical domain is based on only two studies
(Borreani et al., 2014; Holmdahl, Savenstedt & Imoni, 2014) as is the legal domain (Pype et al., 2013;
Anquinet et al., 2015). This could be due to the objectives of the primary study and the questions asked,
and on the other on the focus of the participants on their micro-context. The epidemiological context
domain was not served by any study, probably due to the fact that information concerning
epidemiological context is most likely to be reported in the introduction, method section or in the
discussion. Alternatively, epidemiological studies would not meet the inclusion criteria as these studies
present quantitative data.
The majority of the included studies were adequately reported. Seven studies were inadequately reported
(Harding et al., 2012; Bertero, 2011; Carlsson, 2014; Milbert & Strang, 2000; Pype et al., 2013; Slort et
al., 2011; Wilson et al., 2014), while 34 were adequately reported. Exclusion of these seven studies
would have led to the removal of the categories of “Socio-economic status of Recipients of care“(Wilson
et al., 2014) (socio-cultural domain) and “Tasks, time pressure and workload” (Bertero, 2011; Wilson et
al., 2014) (provider domain). Additionally, the domains “Decision-making concerning sedation”, “Sharing
information” (both legal domain), “Social understanding of health issue “Social support” (both socio-
cultural domain) as well as “Autonomy” (ethical domain) would suffer in terms of their validity since they
would only be based on one study.
136
Methodological limitations
The results of this review are based on comprehensive systematic searches of four databases. Search
filters identifying qualitative research are prone to result in a large number of false positive (Shaw et al.,
2004). Thus, the search strategy would have benefitted from additional search strategies such as hand
searching of all relevant journals, identification of studies citing other studies, as well as contacting
authors of studies that were included into the effectiveness review. All steps of this review have been
undertaken by one co-author only (e.g. title and abstract screening, data extraction, quality assessment)
unless an article was assessed as “unclear” for inclusion when a second author was consulted.
Moreover, our review reflects solely perspectives of European stakeholders in HBPC. This inclusion
criteria, however, facilitates the integration of the review findings with other findings from the
INTEGRATE-HTA project.
Framework synthesis moreover seduces data extractors to make data fit the framework. This could
prevent the formation of new categories outside the a priori framework. However, in our case, the generic
framework is of high granularity. Thus, the formation of sub-categories was of particular interest. For
example, the formation of the subcategory of family in the socio-cultural domain was highly relevant when
considering the context for home-based palliative care.
Reflections on the application of the CICI framework
Application of the framework
The applicability of the CICI framework was relatively good. This might largely be due to the high
granularity of the framework. The application however offered insights into the substructure of the
framework and what should be contained in the single domains. Also, it gave a good insight into the
interconnectedness of domains.
Conversely, the granularity of the framework may have undermined the allocation of data from different
aspects to one specific domain since various possibilities were available. Hence, data were allocated to
more than one domain and flagged to indicate their use in more than one domain. The synthesis involves
the data extraction using an a priori framework that is then adapted by applying secondary thematic
analysis to the evidence (Carroll et al., 2013). Newly emerging subcategories were discussed with all co-
authors. At this stage, links between domains as well as overlaps were discussed and flagged for
analysis. Additionally, two specialists of socio-cultural (KM) and ethical issues (KBL) looked specifically at
the ethical and the socio-cultural domain to check for overlaps between the two domains.
The exercise of allocating the data (verbatim quotations and interpretation of authors) into specific
domains of the CICI framework was perceived to be difficult by the researchers completing data
extraction. This can be partly explained by the different background and expertise of the researchers and
partly by fact that the data is relevant to more than one domain. Hence, the allocation of data to domains
of the CICI framework can be somewhat arbitrary. We can argue that this may not be a problem as long
as all data are reported. However, the domains frame the way the barriers are perceived, and accordingly
where to look for solutions. This is of particular relevance for decision makers.
Conclusion
In HBPC, significant barriers and facilitators to the implementation of this complex intervention were
identified. The provider, the organisation and structure in which they are embedded as well as the micro-
context of the family and home in which a palliative care patient and his family are living were of particular
relevance to the successful delivery of palliative care. Interactions between the single domains were
described in detail, highlighting connecting factors for future interventions.
137
While the patient wishes to receive palliative care at home and also to die there, the delivery of informal
care imposes a burden on the lay caregiver. As informal caregivers are reluctant to reveal their needs
and professional caregivers lack training, resources and experience structural barriers to respond,
informal caregiver’s needs for support fail to be identified and addressed. Furthermore, the access of the
lay caregiver to the health and social system as well as barriers to actually utilising them were eminent
themes in several included studies. In particular, difficulties in navigating the system were identified by
various participants.
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Completing the evidence summary templates
Using a generic template to summarise the major findings in an accessible and user-friendly way,
evidence summaries were developed for:
Effectiveness,
Economics
Socio-cultural aspects
Ethical aspects
Legal aspects
Patient preferences
Moderators of treatment outcome
Context and implementation issues
The evidence summaries were used to reduce the volume of information at the end of step 3 and formed
the basis for the presentation of evidence to the decision making committee in the case study.
Summary of Step 3
Step 3 presented the evidence for each assessment aspect (i.e. effectiveness, economics, socio-cultural,
ethical, legal aspects, patient preferences, moderators of treatment outcome, context and implementation
issues).
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CHAPTER 4
Step 4 – Mapping of the evidence
Overview of Step 4
This chapter presents Step 4 which involves synthesizing the evidence to inform the research question.
This step provides an overview of the HTA results to specifically answer the HTA research question (See
figure 4.1). Step 4 identifies overlaps in the outcomes provided by evidence summaries from different
assessment aspects and to ensure that specific outcomes inform the relevant assessment criterion of the
HTA research question. Accordingly, the evidence for each assessment criterion (such as "acceptability")
is merged from different assessment aspects (such as the ethical, legal and socio-cultural assessment)
that applied different assessment methods. However, after feedback in the meeting, internal and external
review, we subsequently developed the extended logic model to assist decision making. Although the
extended logic model to assist decision making was not tested in this case study, it is included as an
example of how the case study application has assisted the development of the methods in the project.
141
Figure 4.1 Step 4 of the INTEGRATE-HTA Model
(Wahlster al., 2016)
142
Assessment criteria
Using evidence from the evidence summaries to inform the assessment criteria
The overall question for the case study was: “Are reinforced models of home based palliative care
acceptable, feasible, appropriate, meaningful, effective, cost-effective model for providing patient-
centred palliative care [compared to non-reinforced (i.e. ‘usual’) models of home based palliative care] in
adults (defined as those aged 18 years old and over) and their families?
The evidence needed to answer the question was extracted from the evidence summaries and assigned
to the 6 assessment criteria (effectiveness, meaningfulness, acceptability etc.). The structure was created
in cooperation with the researcher completing the assessment of individual aspects of the HTA (i.e.
effectiveness, ethics). This process involved:
The researcher completing the assessment of individual aspects of the HTA (i.e. effectiveness,
ethics) suggested evidence that informed each assessment criterion (such as “vulnerability” as an
ethical outcome for the assessment criterion “meaningfulness”).
A second researcher reviewed the suggestions made to identify overlaps in the evidence (e.g.
autonomy and shared decision-making consists of evidence from the legal, ethical, preferences,
socio-cultural assessment). The researchers ensured that the evidence was used to inform only
one assessment criterion to avoid presenting the same pieces of evidence several times in step 5.
Step 4 involved extracting evidence from the evidence summaries from each assessment aspect (e.g.
effectiveness, ethics, sociocultural etc) and assigning this to the 6 assessment criteria (effectiveness,
meaningfulness, acceptability etc.) (see figure 1). Summary tables of the evidence informing each
assessment criterion were developed in preparation for decision making (see Appendices 40 - 46). Each
table provided a summary of the evidence that informs a specific assessment criterion. In addition, issues
that may influence the internal / external validity of the findings were reported according to the information
provided by the evidence summaries. Evidence from these summary tables were presented to the
decision making committee in the case study. However, after feedback in the meeting, internal and
external review, we subsequently developed the the extended logic model to assist decision making (see
Figure 4.3).
Rapid applicability assessment
To apply the HTA findings to a specific context, a rapid applicability assessment to identify contextual and
implementation barriers and enablers specific to the context can be conducted. This includes the
application of a consultation guide to local experts (see Appendix 47). The consultation guide can be
used by researchers and policy makers as a tool to assess the applicability of HTA findings by retrieving
setting-specific information through the consultation of local experts. Therefore, the rapid applicability
assessment could be performed within this step to assist decision-making in a specific context.
Results
The evidence that was assigned to the 6 assessment criteria according to the process described above is outlined in figure 4.2.
143
Effectiveness
• Caregiver
• Quality of life
• Response Outcomes
• Satisfaction with care
• Psychological health (plus moderators)
• Patients
• Pain
• Symptom control
• Quality of life
• Psycological health
• Hospitalisation
• Response
• Satiscfaction with care
• Death at home (plus moderators)
Cost effectiveness
• Costs per patient
• Resources impact (e.g. Specialist Nurse time)
• Budget impact
Acceptability
• Changing roles and relationship for caregiver(ethics)
• Changing roles and relationship for patients (ethics)
• Autonomy and shared decision-making (Legal, ethics, preferences, socio-cultural)
• Location of death (preferences)
• Preference for survival
Meaningfulness
• Vulnerability (ethics)
• Perceived usefulness and the idea of benefit (socio-cultural)
• Knowledge and understanding of the technology (i.e. home based palliative care; socio-cultural)
• User-professionals-relation and decision making (socio-cultural)
Feasibility
• Results of the qualitative review on context and implementation
Appropriateness
• Access and availability (ethics)
• Voluntariness (ethics)
Figure 4.2 Structure of HTA research question taking the HTA results into account
144
However, after feedback in the meeting, internal and external review, we subsequently developed the
extended logic model to assist decision making (see Figure 4.3). The extended logic model to assist
decision making is based on evidence summaries from the assessment of effectiveness, economics,
acceptability, meaningfulness, feasibility and appropriateness (see figure 4.3). This was not tested in the
case study but is included as an example of how the case study application assisted the development of
the methods in the project.
Construction of the extended logic model to assist decision making
Evidence captured in the evidence reports was used to systematically complete a matrix that captures
interactions and the direction of effects between outcomes. This evidence along with the summary tables
and the specific logic model from step 2 provide the structure for the development of the extended logic
model to assist decision making. The boxes within the extended logic model to assist decision making
such as epidemiology, political context are consistent with the logic model to allow an easy
transformation of the logic model into the extended logic model to assist decision making. However,
whilst the specific logic model in step 2 specifies what evidence is relevant to the HTA research question,
the extended logic model to assist decision making results in a comprehensive, transparent and
integrated illustration of all assessment results. It is a new tool that was developed in this project as
graphical way of informing decision makers about aspects related to the technologies of interest. The
outcomes that were assigned to the assessment criteria of the HTA research question (meaningfulness,
appropriateness etc.) are illustrated within the extended logic model to assist decision making.
How to read the extended logic model to assist decision making
The assessment criteria (effectiveness, meaningfulness etc) are symbol coded. Evidence relating to each
assessment criterion is coded by the same symbol and assigned to either context (legal, ethical and
socio-cultural evidence) or implementation outcomes (effectiveness and cost effectiveness evidence,
patient preferences and moderators of treatment effect). Where an outcome consists of evidence from
the assessment results of more than one assessment aspect (e.g. legal, ethical etc.), the outcome is
assigned to several areas in the extended logic model to assist decision making. For instance, the
outcome “Autonomy and shared decision-making” was located in four different assessment aspects as
the underlying evidence was obtained from the assessment of these four different aspects (Legal, ethics,
patient preferences, socio-cultural aspects).
145
Figure 4.3 The extended logic model to assist decision making showing the assessment results on reinforced models of palliative care (sources of evidence are highlighted by numbers)
Legal
Autonomy and shared
decision-making (Legal,
ethics, preferences)
Context
Socio-economic
Implementation Outcomes
Patients, informal caregivers
Intervention = reinforced home care
Policy
Meeting a society�s demands
Faciliation of deadth at home
Navigation the health care system
Social protection of informal caregver
Funding Financial support of informal caregiver
Availability of resources for care provision
Not being worth the investment
Organisation and structure
Continuty of care
Supervision and guidance
Intervention Theory, aim &
scopeholistic approach to improve quality of life and to enable a good death for patient
aim to allow the patient to be treated for and die at home, if desired
explicit, structured support for the lay caregiver to; alleviate burden due to caregiving
Components
Services addressing physical, psychological, social and spiritual needs of patients
Services explicitly providing psychosocial or psychoeducational support to lay caregiver
Delivery mechanisms
Face-to-face /distant (telephone, online)/mixed
Individual/group/patient-carer dyad/mixed
Delivery agent
Generalist and/or Specialist health and social care professionals
Lay caregivers
Others: Self-care, complementary and alternative therapists, charity workers/volunteers
Execution
Timing, duration and frequency
May commence at any time from diagnosis to end of life and bereavement
Models of transition to palliative care e.g. concurrent palliative and curative care; palliative care upon cessation of curative care
Moderators for
Preferences for
Health outcomes
Economic outcomes:
Geographical
Epidemiological
Main assessment criteria according to the HTA research question
Effectiveness Cost-effectiveness Acceptability Feasibility AppropriatenessMeaningfulness
Caregiver: Psychological health (preferences,
outcomes)
Patients:Death at home
(outcomes, preferences)
Preference for survival
Autonomy and shared decision-making
(Legal, socio-cultural, ethics, preferences)
Location of death
Caregiver
Quality of life
Response Outcomes
Satiscfaction with care
Psychological health (outcomes, preferences)
Patients Pain
Symptom control
Quality of life
Psycological health
Hospitalisation
Response
Satiscfaction with care
Death at home (outcomes, preferences)
7
Budget impact
Resources impact (e.g. Specialist Nurse time)
Costs per patient
Political Socio-cultural
Autonomy and shared decision-making (Legal,
socio-cultural, preferences)
Perceived usefulness and the idea of benefit
Knowledge and understanding of the
technology
User-professionals-relation and decision making
Ethics
Voluntariness
Access and availability to health care (ethics, socio-economic,
geographical, political)
Vulnerability
Autonomy and shared decision-making (Legal, socio-cultural,
ethics, preferences)
Changing roles and relationship for caregiver
Changing roles and relationship for patients
Provider
Characteristics of provider
Team dynamics
Interaction with care recipients
Training needs
Infrastructure
Access and availability to health care
(ethics, socio-economic,
geographical, political)
Access and availability to health care
(ethics, socio-economic,
geographical, political)
Socio-economic status of recipient of care
Economic climate
Access and availability to health care (ethics,
socio-economic, geographical, political)
1
1
1
1
1
1
1
1
1
1
1
1
1
1
2
2
2
2
2
3
3
3 3 33
3
3
3
3
3
Key to numbers assigned within the symbols: Evidence informing the extended logic model resulted from the application of the following guidance: 1 = Lysdahl et al. (2016a) 'Guidance for assessing effectiveness, economic aspects, ethical aspects, socio-cultural aspects and legal aspects in complex technologies. [Online]. Available from: http://www.integrate-hta.eu/downloads/ 2 = Van Hoorn, et al (eds.) (2016a) Guidance for the assessment of treatment moderation and patients’
preferences [Online]. Available from: http://www.integrate-hta.eu/downloads/ 3= Pfadenhauer et al. (2016) Guidance for the Assessment of Context and Implementation in Systematic Reviews and Health Technology Assessments (HTA) of Complex Interventions: The Context and Implementation for Complex Interventions (CICI) Framework [Online]. technology assessments of complex interventions & Rohwer et al. (2016) Guidance: Use of Logic Models in Health Technology Assessments of Complex Interventions [Online]. Available from: http://www.integrate-hta.eu/downloads/
146
Plausibility check by SAPs/HTA researcher
The HTA researchers involved in the project reviewed the assignment of evidence to the assessment
criteria of the HTA research question. They also checked the extended logic model to assist decision
making regarding the plausibility of the illustration. As such the model was iteratively revised.
Results of the rapid applicability assessment
We applied the applicability assessment, which is based on the CICI framework, in three different
countries, Germany, Poland and UK. General findings include that organizational and structural barriers
would need to be considered in all three countries to ensure applicability of rHBPC. The underlying issue
to many of the stated barriers concerning the implementation of rHBPC is the limited availability of
financial resources. Detailed results of the applicability assessment for each of the countries and
methods are listed in Appendix 47.
Summary of Step 4
The processes that were developed to use evidence to inform the assessment criteria have been
outlined. Summary tables informing each assessment criterion assisted in ensuring that the volume of
evidence presented was manageable for decision making. Finally, step 4 resulted in the development of
the extended logic model to assist decision making which provides a graphical illustration of all
assessment results. At this stage, the assessment results are organized in a way that allows their
presentation to decision-making bodies and other stakeholders interested in the results. Furthermore, a
rapid applicability assessment could be conducted to identify contextual barriers and enablers when
applying the HTA findings to a specific setting. The next stage, step 5 describes how the evidence was
processed for decision-making on the HTA research question.
References
LYSDAHL, K.B., BRERETON, L., OORTWIJN, W., MOZYGEMBA, K., REFOLO, P., SACCHINI, D., BRÖNNEKE, J.B., VAN DER WILT, G.J., GERHARDUS, A., HOFMANN, B. (2016b) Guidance to assess ethical aspects. In: LYSDAHL, K.B., MOZYGEMBA, K., BURNS, J., CHILCOTT, J.B., BRÖNNEKE, J.B., HOFMANN, B. (eds.). Guidance for assessing effectiveness, economic aspects, ethical aspects, socio-cultural aspects and legal aspects in complex technologies [Online]. Available from: http://www.integrate-hta.eu/downloads/
PFADENHAUER, L., ROHWER, A., BURNS, J., BOOTH, A., LYSDAHL, K.B., HOFMANN, B., GERHARDUS, A., MOZYGEMBA, K., TUMMERS, M., WAHLSTER, P., REHFUESS, E. (2016) Guidance for the Assessment of Context and Implementation in Health Technology Assessments (HTA) and Systematic Reviews of Complex Interventions: The Context and Implementation of Complex Interventions (CICI) Framework [Online]. Available from: http://www.integrate-hta.eu/downloads/
VAN HOORN, R., KIEVIT, W., BOOTH, A., LYSDAHL, K.B., REFOLO, P., SACCHINI, D., MOZYGEMBA, K., HOFMANN, B., GERHARDUS, A., PFADENHAUER, L., TUMMERS, M., VAN DER WILT, G.J. (2016c) The retrieval and critical appraisal of literature on patient preferences for treatment outcomes. In: VAN HOORN, R., TUMMERS, M., KIEVIT, W., VAN DER WILT, G.J. (eds.). Guidance for the assessment of treatment moderation and patients’ preferences [Online]. Available from: http://www.integrate-hta.eu/downloads/
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CHAPTER 5
Step 5 - HTA decision making
Overview of Step 5
This chapter presents Step 5 which results in the HTA decision making and recommendation (See figure
5.1). Step 5 presents the HTA results from steps 3 and 4 to a decision making committee using a tool to
structure a deliberative discussion. This step enables unanswered issues (perhaps because the issues
were not raised yet) and uncertainty surrounding the decision to be discussed by decision makers. The
discussion of the decision committee represents the final validation of the process before making the final
HTA decision/recommendation.
This chapter describes a ‘mock’ decision making meeting that was completed as part of Step 5 of the
case study. The ‘mock’ meeting simulated a decision making meeting although there was no intention to
actually make and implement a decision because this specific HTA was requested not by service
commissioners. The intention was to weight and score all of the evidence per assessment criterion which
would identify evidence gaps before making the final (‘mock’) decision. Unfortunately, time restraints
meant that evidence was presented for only two of the assessment criteria and a final decision was not
made. However, valuable lessons were learned that enabled development of the INTEGRATE-HTA
model.
148
Figure 5.1 Step 5 of the INTEGRATE-HTA Model
(Wahlster et al., 2016)
149
Stakeholders involved in the final meeting:
A stakeholder group’ most of whom were professionals who are regularly involved in commissioning end
of life services in England, participated in Step 5. In total, 13 participants assigned to different
stakeholder groups attended the meeting. 2 lay people (one male, one female, both former caregivers)
and 11 health care commissioners from the local end-of-life committee with different backgrounds (e.g.
physicians, service commissioners, former caregivers) participated in one final meeting. The latter
consisted of 3 physicians, 4 nurses and 4 with other backgrounds. 10 females (77%) and 3 males (23%)
attended the meeting. The group weighted the relevance of assessment criteria (see appendix 47).
Relevant permissions were gained from the NHS and the board of trustees of St Luke’s Hospice prior to
the meeting taking place.
Presentation of the HTA results to a decision committee
The final meeting aimed to enable a detailed evaluation of benefits and drawbacks of reinforced palliative
home care. This was based on the evidence reports (as the extended logic model to assist decision
making was not available in the final format at the time of the ‘mock’ decision making meeting).
Selecting a decision support tool to structure a deliberative discussion in the meeting
As outlined in the integrated assessment of complex health technologies guidance21, different
approaches to decision making are available. Multiple Criteria Decision Analysis (MCDA) is one such
approach. As highlighted in several studies, MCDA can support decision-making in HTA (Wahlster et al
2016). We selected a simple MCDA method that was based on the direct weighting technique for
practical reasons (i.e. due to the limited time of participants involved in this exercise).
The ‘mock’ decision making meeting
The intention was to weight and score all of the evidence per assessment criterion which would identify
evidence gaps before making the final (‘mock’) decision. The ‘mock’ decision making meeting was audio
recorded, providing a record of comments made and enabling the researchers to reflect on the issues
raised. The meeting started with a short introduction of the INTEGRATE-HTA project. Background
information on the project and the case study, including a definition of reinforced and non-reinforced
models of home based palliative care was presented. Participants were given brief information about
evidence hierarchies and internal and external validity to ensure all participants had some understanding
of these concepts before rating the HTA evidence. Stakeholders then took part in:
1) General weighting of assessment criteria and sub criteria of the HTA research question (i.e.
effectiveness, acceptability, feasibility etc.)
2) Scoring and discussion of HTA results
Unfortunately, time restraints meant that evidence was presented for only two of the six assessment
criteria and a final decision was not made.
General weighting of the assessment criteria of the HTA research question
In a written exercise, each participant was asked to distribute 100 points across the six criteria in the
main HTA research question (i.e. effectiveness acceptability feasibility etc.) to weight these criteria.
The six assessment criteria were weighted independently from the technology of interest (in our case
reinforced models of home based palliative care). The allocation of points was based on a generic
description of the assessment criteria.
21
Wahlster P, Brereton L, Burns J et al (2016) Integrated assessment of complex health technologies – The INTEGRATE-HTA model [Online]. Available from: http://www.integrate-hta.eu/downloads/
150
Participants then allocated a further 100 points to the sub-criteria of each of the main assessment criteria
e.g. effectiveness for patients and effectiveness for caregiver as two separate criteria. The task was
repeated for the sub-criteria of each of the assessment criterion. Descriptive statistics were applied and
mean standard deviations (SD) calculated. The results of this exercise identified ‘weights’ for each
criteria. These ‘weights’ represent the overall importance of the different assessment criteria to
participants involved in the meeting.
Doing this illustrated the stakeholders’ preferences for different assessment criteria of the HTA research
question and ratings of the underlying evidence. Results on preferences for the different assessment
criteria were directly presented back to the committee and informed a final deliberative discussion.
Presentation and scoring of evidence and identification of evidence gaps
Having completed the weighting exercise, the performance of reinforced models of home based palliative
care was scored against each assessment criterion. Participants provided a ‘performance score’ for each
of the six criteria (i.e. effectiveness acceptability feasibility etc.) for reinforced and non-reinforced models
of home based palliative care using the underlying available evidence from the HTA case study. As this
exercise was not completed for all of the assessment criteria, details of the scoring exercise are provided
in appendix 48. In addition, participants deliberatively discussed the evidence presented in the meeting.
Some of the key issues raised in this discussion are presented below.
Results
The results of the weighting exercise are presented here. However, the scoring exercise could not be
completed for all of the evidence. Those available are presented in appendix 49. The discussion raised a
number of issues that subsequently resulted in the development of the extended logic model to assist
decision making as an alternative way of presenting integrated evidence to decision makers.
Participants’ weighting of the main assessment criteria
Regarding weights provided by meeting participants, figure 5.2 shows that the most important criteria
were “Effectiveness” (mean weight 29.9 points or 29.9 % [Standard deviation (SD) 12.1%]),
“Acceptability” (16.3% [SD 9.4%]) and “Meaningfulness” (15.8% [SD 6.7%]). Least important mean
criteria were “Feasibility” (10.8% [SD 7.6%]), “Appropriateness” (13.1% [SD 12.0%]) and “Cost-
effectiveness” (14% [SD 9.4%].
The largest consensus across participants on the weights for the main criteria were observed for
‘‘Meaningfulness’’ (SD 6.7%) and ‘‘Feasibility’’ (SD 7.6%). The largest variations in main weights were
observed for the criteria “Effectiveness” (SD 12.1%) and ‘‘Appropriateness” (SD 12%), “Acceptability” and
“Cost-Effectiveness” (both SD 9.4%) (See Figure 5.2).
Figure 5.2 Mean weights of main assessment criteria in percent. Participants distributed weights (100 points) across the main assessment criteria.
151
Participants’ weights on sub assessment criteria
This section reports on how participants allocated 100 points to the sub criteria assigned to each main
assessment criterion.
Effectiveness:
The main criterion “Effectiveness” was divided into two sub criterion; effectiveness for patients and
effectiveness for caregiver. . Firstly, participants weighted effectiveness for patients as slightly more
important (57.5% [SD 5.8]) than effectiveness for caregiver (42.3% [SD 5.8]) (see Figure 5.3). This
provides some insight into the priorities of the decision making committee and possibly the relevance of
the research question about reinforced models (an intervention primarily aimed at supporting care givers)
for decision makers.
Figure 5.3 Relative weights for effectiveness for caregiver versus effectiveness for patients.
Regarding the four outcomes on effectiveness for caregivers, the most important outcomes for
participants were “Quality of life” (28.8% [SD: 15.4]) and “Satisfaction with care” (28.5% [SD: 11.4]). The
least important outcomes were the “Response” (21.9% [SD: 7.8]) and “Psychological Health” (20.8% [SD:
7.0]). Still, the comparable weights of these four outcomes indicate an equal importance.
The effectiveness for patients consist of 9 outcome parameters with the most important parameters
“Quality of life” (18.2% [SD13.2]) and “Pain” (17.5% [SD: 9.4]). The least important parameters were
29.9 14.0 16.3 15.8 13.1 10.8
0% 10% 20% 30% 40% 50% 60% 70% 80% 90% 100%
Mean Weights Main Criteria
Effectiveness E
Cost EffectivenessCEAcceptability AC
Meaningfulness M
Appropriateness AP
Feasability F
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“Hospitalization (7.6% [SD: 4.6]) and “Death at home” (9.0% [SD: 3.1]). This could hint at the priority of
traditional outcome parameters such as “Pain” and “Quality of life” for participants.
“Appropriateness”.
The main criterion “Appropriateness” was divided into two sub criteria “Access and availability” (53.1%
[SD: 4.8]) and “Voluntariness” (46.9% [SD: 4.8]) which both receive nearly equal weights.
Meaningfulness:
The sub criterion “User-professionals relationship and decision-making” (38.5% [SD 13.3]) was the most
important criterion compared to the 3 other sub criteria “Vulnerability” (21.9% [SD: 12.0], “Perceived
usefulness and the idea of benefit” (21.9% [SD: 10.91, “Knowledge and understanding of the technology”
(17.7% [SD 8.8]). These weights indicate pivotal importance of the relationship between professionals,
patients and carers and the implications for decision-making in palliative care.
Acceptability:
The most important criteria were “Autonomy and shared decision-making” (24.6% [9.0]) and the Location
of death (23.1% [14.4]). The least important criterion was “Survival (12.7% [9.7]) which seems logical
considering the essence of palliative care is not meant to prolong survival in the first place (compared to
curative care).
Cost-effectiveness:
Cost-effectiveness reported 3 sub criteria. “Budget Impact” (39.8% [SD: 22.8] and “Resource impact”
(36.8% [SD 18.1]) received higher importance than the “Costs per patient” (23.4% [11.7]). Accordingly,
one conclusion could be that costs for individuals do not matter if the number of patients is small and
overall costs do not significantly affect the overall budget.
Feasibility:
No sub criteria were presented for feasibility as the evidence summaries were not available at the time of
the meeting.
Participants’ discussion on the HTA results of the case study
Participants also provided quantitative scores on the HTA results using scales from -5 to +5 (details are
provided in appendix 49). However, the scoring exercise was difficult for participants for several reasons,
which are documented with reference to each step of the INTEGRATE-HTA model below. Decision-
makers raised open questions about the scoring exercise and methods applied in the case study
tthroughout the final discussion. Although time for discussion was limited in the mock decision making
meeting, decision-makers raised a number of key issues which are outlined below. After the meeting, we
were able to assign the issues to each step of the INTEGRATE-HTA Model as follows.
Step 1 Definition of the HTA objective and technology
Value of the HTA objective and focus on models of palliative care.
The focus on models of service delivery as the technology of interest is appropriate. Participants
indicated that commissioners valued the opportunity to consider evidence that draws on more than cost
and clinical effectiveness evidence, such as evidence related to “Appropriateness” and “Meaningfulness”,
which is not normally available to them. Accordingly, the HTA objective including the technology of
interest and the assessment criteria were appreciated by the mock decision panel.
Step 2 Creation of a logic model to define evidence needs
Need for clear definition of reinforced care - comparing apples and pears
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Several comments indicated that decision makers reported some difficulties in identifying differences
between reinforced and non-reinforced models of palliative home based palliative care. Some
participants thought that caregiver support is already a component of current home care provision. The
initial question raised by a participant was: “Do we not already do that? We do have organized
counselling sessions and we do it…”. This led to an important discussion amongst participants during the
meeting that clarified the difference between reinforced and non-reinforced home care. A participant
concluded: “The difference between reinforced home based palliative care as we provide it and the
studies is that this [the reinforced element of informal caregiver support] is a much, much more formal
process. So it’s a program of education, an intervention directed at it, which is much more structured than
what we provide at the moment. And I think that is … because we routinely view the care that we deliver
as care we deliver both to patients and their families or carers and we would routinely be doing our best
to reinforce and support families in emergency care planning and that would be a routine part of our work.
But I see what this project is looking at are much more formal approaches. So, a set program of training
and education, over a defined period of time with defined outcomes that you can see very clearly: this is
what it is. And we don’t take it to that extent.“ This statement clearly acknowledges that reinforced models
have an extra element of caregiver support which is explicit and embedded in a structured program.
Indeed, the discussion raises an important issues influencing this case study, notably regarding
stakeholders’ understanding of a) reinforced home care and how this differs from non-reinforced home
care and b) the level of carer support provided.
Despite this clarification and brief explanations about the interventions used in reinforced home based
palliative care some decision-makers struggled to understand what the reinforced interventions were that
they were making decisions about. Indeed, reinforced home based palliative care describes a range of
interventions designed to support informal caregivers. Some of these interventions are psycho-social,
some of them are preparation of care-giving and some of them are counseling. Ideally, the health
technology assessment of reinforced care would address all of the different intervention components
included in reinforced models of home based palliative care. However, this was not possible due to the
limited amount of evidence that exists about reinforced models of home based palliative care. Hence, we
could only provide evidence on the overall effect of reinforced home care. As pointed out by one
participant, “we know in some trials, palliative care is one clinic appointment – [it is] hard to compare this
to a psychological program or counseling.” Another decision-maker stated “again [it is] difficult to tell what
[the] intention was – [we] can be comparing apples and pears”. The comparison between reinforced and.
non-reinforced palliative home care was based on a Cochrane Review on palliative care that was
identified by HTA researchers. This could indicate a gap between the perception and categorization of
palliative care in the HTA community and that of the palliative care community.
Step 3 Evidence collection and preparation
External validity, evidence gaps and the need for a comparator
External Validity
Participants’ comments hint at the importance of external validity when it comes to complex interventions
such as models of home based palliative care. Participants were specifically concerned about the
external validity of the evidence, mostly related to studies from other countries. For instance, a participant
highlighted that “palliative care often means different things” in the US as there are “Significant cultural
and healthcare organization differences between UK and USA having a big impact here.”
At one point, participants discussed the funding system for palliative care in Australia in an attempt to
determine the transferability of Australian study results to the UK context. A general comment was that
“different things will work in different cultures, personalities etc. and [it is] difficult to generalize across a
country.”
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Evidence gaps
Decision makers consider the evidence gaps to be significant. A general comment of several participants
at various points in the meeting was that judgments of evidence were not possible based on the available
data.
Regarding the qualitative evidence presented about “changing roles and relationships”, a decision-maker
commented that “one of the things that we talked time and time again that the number of people going
into a house has a negative impact on carers because the struggle to understand what everybody’s
responsibilities are. So, there’s a risk that this model could introduce another person and that’s not on
here as a consequence.”
The need for a clear comparator
During the decision making meeting, we discussed evidence regarding the two main assessment criteria
of “Effectiveness” and “Acceptability”. Whereas quantitative evidence mainly informs “Effectiveness”
mostly qualitative evidence informs “Acceptability” decision making criteria. A key difficulty for decision
makers was that the qualitative evidence does not allow them to easily differentiate between the
reinforced and non-reinforced models of home based palliative care. Some participants struggled to
make a decision about the acceptability of reinforced and non-reinforced models of home based palliative
care because, as highlighted by a decision-maker, it seemed that: “All the [qualitative] evidence speaks to
a standard home care model so it is not possible to say one is better than the other.”
When judging evidence about “Changing roles and relationships for caregivers”, one participant said:
“there is not enough information to base a judgment on this based on pure reasoning. We can’t say if
reinforced models might make things worse. So I really can’t say. Even from an ethical perspective it is
difficult to say.” Another participant agreed: “We just don’t know if the reinforced model might actually
make things worse(…) you can say, my instinct is that it may (…).You just don’t know. Because it might
make everything worse.”
Another decision maker was undecided about the direction of effect as reinforced care “may help some
people and give them confidence but may provide an added burden to others who may feel more
responsibility for the care given and less likely to ask for help.” Another participant agreed as the impact
on roles and relationships is “highly variable from person to person. Being aware of different responses
can help judge the situation, but cannot predict the response.” With regard to “patient autonomy”, a
participant indicated that in reinforced home based palliative care there is the “potential for patients to feel
less autonomous as more attention on the carer role is raised.”
In contrast, some participants were convinced that reinforced care improves the role and relationship for
caregivers. One felt that “improved preparedness, training, confidence etc. will outweigh standard care”.
Step 4 Evidence processing and integration
Overlaps between some categories in the decision making criteria
Interactions between different assessment aspects
Decision-makers’ feedback indicated overlaps between some categories of the decision making criteria.
This is important as assessment criteria should be technically robust and avoid potential overlaps.
For MCDA, every criterion should focus on a single aspect of the decision to avoid double-counting.
Double-counting means that the overlapping pieces of evidence between criteria can contribute several
times to the decision. During the weighting of the main assessment criteria, one participant asked
whether “for it to be meaningful, it must be acceptable?” This question is referring back to step 1 as the
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assessment criteria of the HTA research question were determined in this step without sufficient
consideration of these overlaps.
On the level of sub criteria, this was also a relevant issue. During the weighting of patients’ effectiveness
a participant assumed that “good pain control/symptom control is also measured in Quality of Life”.
Another statement was made during the weighting of “Appropriateness” sub criteria suggesting that all
criteria were “interrelated”. Due to limited time, it was not possible to validate the structure of the criteria
with external stakeholders. A Delphi panel would have potentially been an appropriate method to
generate consensus on the structure before its application.
Step 5 Decision-making
Final decision
Lack of time was a major obstacle to the discussion of issues and final decision making in the meeting.
Arrangements were made to meet with the group for 2.5 hours. However, communication difficulties
regarding arrangements led to a delay in commencing the meeting, which coupled with competing
commitments for busy professionals meant that the meeting was reduced to 1.5 hours on the day. The
final decision on the value of reinforced palliative care could not be completed. As outlined above, this
resulted from a) difficulties differentiating between reinforced and non-reinforced home based palliative
care b) participations’ view that evidence is not sufficient to make a decision c) difficulty making decisions
based on reasoning about possible effects as many variations are possible d) limited time to finalize
ratings in the mock decision making meeting and e) only part of the evidence (effectiveness,
acceptability) could be addressed. See appendix 50 for a summary of feedback from stakeholders on
their involvement in the final meeting.
The next chapter outlines some of the key lessons learned from all the elements of the case study and reflects on the strengths and limitations of the approaches that were piloted.
References
WAHLSTER, P., BRERETON, L., BURNS, J., HOFMANN, B., MOZYGEMBA, K., OORTWIJN, W., PFADENHAUER, L., POLUS, S., REHFUESS, E., SCHILLING, I., VAN HOORN, R., VAN DER WILT, G.J., BALTUSSEN, R., GERHARDUS, A. (2016) Guidance on the integrated assessment of complex health technologies - The INTEGRATE-HTA Model [Online]. Available from: http://www.integrate-hta.eu/downloads/
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CHAPTER 6
Discussion and Conclusions
Overview
This chapter draws together the lessons learned from the case study for both HTA research and palliative
care as well as the key findings from the case study and reflections on the extent to which the aims of the
case study have been achieved. The chapter also outlines the strengths and limitations of the case study
before recommendations for future primary research, HTA and practice are made.
Lessons Learned
The lessons learned have been considered with reference to a number of key issues, notably complexity,
stakeholder involvement in HTA, communication with stakeholders, the evidence base for palliative care,
the value of the methods developed and integration.
1. Complexity:
Palliative care demonstrates all of the characteristics of complexity. Hence, rHBPC and HBPC are good
examples of complex health technologies, although considerable similarity exists between these two
models of palliative care. The complexity characteristics of rHBPC and HBPC have been identified,
adding to our understanding of complexity within home based models of palliative care. This increased
understanding has enabled us to consider the implications for HTA methods (e.g. using harvest plots
instead of meta-analysis for the effectiveness assessment and selecting an appropriate tool for the
ethical assessment).
The holistic philosophy of palliative care means informal caregivers are sometimes targeted by non-
reinforced models. Additionally, reinforced carer support is not a single well defined intervention, with
some interventions being similar to those that may be provided within non-reinforced models (e.g.
education). As a result, locally, nationally and internationally, stakeholders do not have a shared
understanding of palliative care, rHBPC or HBPC. Practitioners and other stakeholders are not familiar
with the term ‘reinforced’ models of home based palliative care and terms such as ‘palliative care’ have
different meanings for stakeholders (sometimes including professionals working in the same team). This
indeterminate phenomena enhances complexity and means that stakeholders have difficulty determining
whether interventions are reinforced or not. Hence, the complexity of interventions and similarities with
comparators presents methodological problems in terms of HTA analysis and hindered comparisons of
the two models of home based palliative care. For example, stakeholders in the sociocultural assessment
were unfamiliar with the term ‘reinforced’ care and the similarities in rHBPC and HBPC posed challenges
in determining model type in the effectiveness assessment and qualitative review of enablers and barriers
of context and implementation of home-based palliative care services. Clarity in definitions and key
concepts is important from the outset of an HTA if stakeholders are to have a shared understanding of
the technology being assessed.
2. Stakeholder involvement
Stakeholder, including patient and public, involvement in HTA is expected internationally but
understandings of what this means and the most appropriate methods of undertaking this vary across
countries. Irrespective of the methods used for stakeholder involvement in the case study, there were
some similarities in the important issues identified across the seven countries that took part in the project.
Both lay and professional stakeholder involvement is a vital and valuable resource in HTA as
demonstrated in a number of ways throughout this case study. For example stakeholders identified
important issues that assist in identifying the topic for the palliative care case study.
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They also provided insights that have assisted economic modelling (where their input was essential due
to the limited economic data available within the literature) and the sociocultural assessment.
Furthermore, stakeholders the interpretation of the assessment findings within the effectiveness gap
analysis and the assessment of preferences and moderators for treatment outcomes. As such,
stakeholders provide valuable insights that complement more traditional forms of evidence. Stakeholders
have also given expert insight into applying the HTA findings to a more specific setting, introducing a
simple and rapid way of assessing the applicability of an intervention assessed in the HTA.
The involvement of decision makers with a role in end of life service commissioning was very much
appreciated as their input helped to identify the most important assessment and sub assessment criteria.
The difficulties experienced in the scoring of the HTA results assisted revision of the methods used in this
phase. The discussion raised several key issues of importance. Not only did it confirm the lack of
understanding of the term ‘reinforced’ models of home based palliative care amongst stakeholders
working in the field, it also highlighted the lack of clarity about the difference between rHBPC and HBPC.
The involvement of the decision making committee provided extremely valuable feedback on the
usefulness of the methods used in the decision making meeting, which have subsequently been revised.
Stakeholders have been successfully involved in the case study using a variety of consultation methods,
including face-face, telephone or Skype individual or group meetings. Work with stakeholders has also
taken place in face-face workshops and via email. Interactive forms of communication with stakeholders
can be the most productive as they provide flexible and responsive opportunities to develop shared
understanding of technologies through discussion.
Stakeholder advisory panels should be adequately resourced in terms manpower, time and finance to
ensure realistic expectations of what these can achieve. Within the case study, it was not always possible
to gain NHS permissions required to include all potentially relevant stakeholders due to time restrictions.
Stakeholders are often extremely busy people who have difficulty finding the time to contribute to HTA. A
key issue to consider is the workload involved in assisting on the project. A key to ameliorate the
workload issue for professionals is ensuring that the task has relevance to them. Therefore, demands on
stakeholders needs to be realistic (in terms of time, commitment and activity required). Consideration
should be given to the feasibility and practicality of activities involving stakeholders.
HTA researchers should aim to include a variety of stakeholders and make best use of stakeholder
involvement by sharing the information provided to enable its wider use in the assessment of several
aspects of HTA (e.g. effectiveness, ethics, socio-cultural issues). This will increase heterogeneity in
stakeholder consultation which assists in identifying heterogeneity within the element being assessed.
Stakeholders were enthusiastic and willing to dedicate time to share their experience for all assessment
aspects (i.e. effectiveness, patient preferences and moderators of treatment outcome etc) and the
conduct of palliative care research. They also commented that they found the experience enjoyable,
rewarding (for example feeling valued) and informative (for example, feedback that presentation of
evidence using the harvest plots is a useful and clear way of organizing the complex evidence).
Stakeholder involvement may require the investment of significant amount of time and energy to organize
(depending on the circumstances, stakeholders and activity). In addition to arranging a mutually
convenient time for stakeholder involvement, researchers may need to prepare lay summaries,
refreshments and payment of expenses or reimbursement for the individual’s time. However, successful
stakeholder involvement may have mutual benefits. The additional workload incurred by the HTA
research team due to the involvement of stakeholders can be considered minor compared to the
information gained from this process. Not only is stakeholder involvement important for ethical reasons as
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it provides a ‘voice’ for the end users of HTA, the added value of stakeholder consultation lies in
stakeholders’ ability to:
provide advice / information that enabled us to identify and confirm that similar key issues exist
with regard to palliative care across countries involved in the INTEGRATE-HTA project (see
chapter 2)
provide quick access to relevant information based on experiential knowledge (i.e. colloquial
evidence (Sharma et al 2015) in the absence of other types of evidence (e.g. in the economic
modeling or for an applicability assessment of the HTA findings) or to supplement existing
evidence (e.g. in the gap analysis for the effectiveness assessment )
make links between issues based on experiential knowledge (e.g. in the assessment of patient
preferences and moderators of treatment effect)
Add credibility to the findings and potentially enhance the validity and reliability of the work
completed
Confirm the relevance of findings
Some concern was expressed that asking SAPs to complete background forms and feedback
forms increased the burden for them but the majority were completed and provided useful
information both on who the panel members were and on their perspectives on involvement.
Our experience of working with a wide range of stakeholders using a variety of methods in HTA may be
transferable to public involvement in service development, although this needs confirmation through such
activity.
3. Communication with stakeholders:
Stakeholder communication prior to any activity should be clear and concise with regard to the
purpose of their involvement and the task. Information should be sent in advance of any meeting
as stakeholders reported that this assisted them in preparing for their involvement in the project.
Stakeholders reported that the size and composition of the stakeholder group influences the level
of discussion in meetings, as does the way that the meeting is facilitated.
Stakeholders reported that communication with others was beneficial in terms of learning about
MDT roles and assisting them to consider their role as a mentor to students.
Including specific stakeholders in advisory meetings at any point in the HTA process may enable
more in-depth discussions of particular issues. For example, the inclusion of professionals with
knowledge of issues relating to informal caregivers assisted gap analysis in the effectiveness
assessment.
Researchers should engage in open discussion with stakeholders as part of any advisory meeting
because stakeholders can provide insights into issues that researchers have not previously
considered. For example, in the assessment of patient preferences and moderators of treatment
outcome, the advisory panel pointed out the importance of individualized care.
Stakeholders can also assist in identifying factors that may affect the applicability of an
intervention in a specific setting, which researchers as well as policy makers may not be able to
identify.
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4. The evidence base for palliative care:
Although stakeholders from four countries involved in scoping identified the need to increase home based
palliative care, little volume of evidence exists with regard to rHBPC and HBPC. Poor reporting about the
components of rHBPC and HBPC hindered comparisons of the interventions, exacerbating limitations
relating to volume of evidence. Furthermore, the limited time available for the application of the
methodological guidances in the case study also impaired full identification of the evidence base. These
issues subsequently limited the findings and conclusions that can be drawn for palliative care from this
case study.
None-the-less, the case study has drawn together knowledge from a variety of assessment
aspects (ie effectiveness, costs, ethical, legal and socio-cultural) about home based palliative
care. This assists readers to gain an holistic perspective of issues associated with home based
palliative care, which to our knowledge have not been drawn together in an integrated
assessment before.
5. The value of the methods developed:
The case study found that the individual concepts and methods developed within the project had value in
their application. In some cases, the application of methods enabled further development of the methods.
For example, some amendments were made to the methods used in the ‘mock’ decision making meeting
as there was recognition of the need to provide decision makers with a more integrated summary of the
evidence in a manner that stimulated facilitates discussion of the HTA results. These methods were
successfully applied within the case study and a number of lessons were learned. For example, the
Harvest plots indicated that reinforced home-based palliative care interventions were neither significantly
better nor worse than non-reinforced home-based care with respect to patient outcomes. Furthermore,
there were no effects across caregiver outcomes for the majority of interventions. The cost impact results
lend some provisional support to the addition of reinforced carer interventions in home-based palliative
care. Most patients prefer quality rather than quantity of life and to die at home.
6. Integration
The INTEGRATE-HTA model provided a framework for integration within the case study. In addition to
this formal structure, other mechanisms can also assist integration. For example, the CICI framework and
the logic model are highly interlinked with the CICI framework forming one component of the a priori logic
model. The logic model underpinned some assessments (e.g. the effectiveness and economic
assessments). The formation and the adaptation of the CICI framework for use in the qualitative review
benefitted from the input of other assessment aspects, in particular in terms of ethics and socio-cultural
issues in health technology assessments. Ideally, the qualitative review would have been triangulated
with the findings of the effectiveness review; however, this was not possible due to time constraints.
Finally, the CICI framework as adapted to palliative care formed a component of the evidence model that
has been developed for step 4 of the INTEGRATE-HTA model. The CICI framework was also used in
Step 4 to assess the applicability of the reinforced home-based palliative care model in specific settings.
Integration was also assisted by the shared steps in the methods used in assessment. For example, the
assessment of complexity is a shared initial step of the ethical and sociocultural assessments. The use of
stakeholders across more than one assessment element also assists integration (e.g. sharing
stakeholder information about complexity in the economics and ethics assessment). Drawing together
evidence from the literature and stakeholders’ experience of palliative care also assists integration.
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To further improve integration of the different assessment aspects in HTA, consideration should be given
to the assessment criteria and collection of evidence. Although the assessment criteria were identified
and defined at the outset of the case study, identifying the most important assessment criteria at the
outset of an HTA is valuable in assisting decisions about how to integrate subsequent evidence
collection.
Secondly, use of the guidance on sociocultural issues, ethics, patient characteristics and context and
implementation produced findings that showed considerable overlap (e.g. autonomy, shared decision
making).These guidances need to be better attuned in order to prevent this.
The application of the guidance to assess patient preferences and moderators of treatment outcomes
suggests that links exist between other research areas (e.g. cultural differences in end-of-life decisions
require the integration of evidence from the socio-cultural and patient preferences). Integration with
evaluations of costs and effects is needed in order to identify valuable technologies in view of patient
preferences and moderators. Hence, further challenges exist in integrating factual evidence (e.g.
evidence about costs) with values (e.g. evidence from ethical assessment).
Decision makers themselves highlighted the value of considering wider aspects of a complex technology
in the decision making process but acknowledged that time and resources often limited their ability to
consider more than effectiveness and economic evidence. The value of developing HTA methods that
take account of more than effectiveness and economic evidence has some value for decision makers as
they have difficulty accessing a wider range of evidence and, as one member of the EoL commissioning
group pointed out, acknowledging patient preferences is fundamental to the underlying philosophy of
palliative care.
The implications of the findings from the case study
The implications of the findings are presented with reference to the case study aims – i.e. to:
1) Show the feasibility and value of applying the concepts and methods developed within the
INTEGRATE-HTA project to an integrated assessment of reinforced and non-reinforced models of
home based palliative care as a specific example of a complex intervention.
2) Provide a synthesis of a broad range of patient-centred and integrated evidence within a demonstration HTA’ of reinforced and non-reinforced home based palliative care that can be used by those commissioning and developing palliative care services to support holistic, patient-centred evidence-based decision-making in a complex field which requires a shared understanding of the issues involved.
The feasibility and value of applying HTA approaches developed within the INTEGRATE-HTA project
As the results of chapter 3, 4 and 5 indicate, some of the concepts and methods developed in the
INTEGRATE-HTA project have been applied to an assessment of rHBPC and HBPC in this
‘demonstration HTA’. From the outset of the project, the intention was to demonstrate some of the many
concepts and methods developed in the project as we recognised that concurrent development of the
INTEGRATE-HTA concepts and methods and the case study meant that time and resource availability
would not allow completion of a full HTA.
Lack of time was one obstacle in the application of some guidance (e.g. the sociocultural assessment
and the final decision making meeting). The time available was too short to gain NHS approvals to
access specific stakeholders to participate in the sociocultural assessment. Time was also a major
obstacle to the presentation and discussion of the findings to the decision makers in the final decision
making meeting.
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As a result, decision makers did not draw conclusions or make recommendations in the final mock
decision making meeting. The limited time also meant that it was not possible to test all of the steps in all
of the methodological guidances developed within the INTEGRATE-HTA guidances. However, the issues
encountered relating to time might not be insurmountable in a real HTA given sufficient time and
resources.
A number of shared difficulties were encountered in the assessments, primarily resulting from similarities
between rHBPC and HBPC. These similarities, compounded by poor descriptions of both intervention
and comparator models within the evidence base created uncertainty for researchers trying to
differentiate between rHBPC and HBPC for assessment using evidence review. These difficulties were
compounded by the heterogenous nature of rHBPC and stakeholders’ lack of familiarity with the term
‘reinforced’ palliative care, which resulted in lack of clarity and shared understandings of rHBPC and
HBPC. The limited amount and quality of evidence that was available about rHBPC and HBPC impeded
the conclusions that can be drawn from the application of the assessment methods applied in this case
study. As such, careful interpretation is required when considering the case study results.
Despite these shared difficulties, that undermined development of an integrated HTA, the case study has
demonstrated use of the concepts and methods developed within the INTEGRATE-HTA project,
suggesting that these are feasible and a potentially valuable addition to HTA methods. For example, the
ethical and sociocultural assessments and the economic assessment enabled the complexity
characteristics of interventions and the palliative care system to be identified respectively. Additionally,
the methods used in the effectiveness assessment enable the graphic display of results relating to
outcomes measured which can be considered with reference to the moderators and patient preferences.
Advances were made in terms of developing an integrated approach to HTA, not only through the
inclusion of the assessment of ethical, legal and sociocultural aspects of rHBPC and HBPC that are less
frequently completed, but also through the development of frameworks and process models. The CICI
framework has demonstrable ability to be incorporated into a logic model, used as the framework for the
best fit synthesis and was also included in the INTEGRATE-HTA model, demonstrating its versatility as a
framework suitable for use in assessing complex technologies. The INTEGRATE-HTA model provides a
useful step based structure basis for the future development of integrated HTA as it can serve to organize
the use of a diversity of methods within a single unifying framework. Whilst the case study related to
rHBPC and HBPC as one example of a complex health technology, the successful application of the
concepts and methods developed within the INTEGRATE-HTA project suggests that scope exists to
apply these in the assessment of other complex technologies.
The case study highlights the importance of integration from the very beginning of the HTA. The definition
of the HTA research question, including the assessment criteria, needs particular attention as the
question guides the assessment methods applied in all subsequent steps. The assessment criteria and
process was not made explicit at this early stage, making integration and assessment more difficult after
the identification and synthesis of evidence. Despite these difficulties, it was possible to integrate
stakeholder consultation throughout all stages of the HTA process.
The value of the concepts and methods applied to assess each aspect of palliative care was considered
in chapter 3.
Effectiveness The case study has indicated the potential exploratory/explanatory value of seeking
stakeholder views on the effectiveness evidence summarised using Harvest plots. The combination of
methods providing new insights into the findings from an effectiveness assessment.
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Economics In the absence of evidence to inform an economic analysis, the methods used in the case
study suggest that stakeholders may have a key role to play in providing a description of the system and
information that can assist economic modelling in complex situations.
Ethics The application of the ethical guidance did not provide evidence that allows for drawing any firm
conclusions about moral relevant differences between reinforced and non-reinforced home-based
palliative. However, the application highlighted complexity aspects of HBPC relevant for the ethical
analysis, and identified a range of ethical issues involved. The case study also added arguments for an
integrative approach to HTA, and provided valuable feed-back for improvement of the guidance.
Socio-cultural aspects Heterogenous stakeholder involvement is important in the sociocultural
assessment. The case study indicates that the methods developed provide a systematic approach to
completing sociocultural assessments in HTA. Although the assessment clearly demonstrates overlaps
with the results from the ethical assessment and the qualitative systematic review of rHBPC and HBPC,
use of Cultural Theory within the sociocultural assessment provides a unique perspective for
respondents.
Legal issues Application of the guidance assisted in identifying legal issues of relevance to reinforced
models of home-based palliative care. However, the guidance does not allow any in-depths analysis of
the affected legal issues. Moreover use of the guidance does not always produce clear and easy to
implement results. Hence, the involvement of a legal representative may still be required
Patient-specific moderators of treatment and patient preferences The methods applied within the
case study successfully identified several moderators and patient preferences for treatment outcome
related to (reinforced) models of home based palliative care. Lay and professional stakeholders in an
advisory panel supported these findings and also identified additional factors that were important in their
view. This highlights the importance of drawing together published evidence and stakeholders’
experiences to provide optimal care for home-based palliative care patients and improve personalised
healthcare.
Context and implementation The Context and Implementation for Complex Intervention (CICI)
framework successfully informed the development of the logic model that has been applied in the case
study. This assists in the development of a greater understanding of complexity and complex systems.
The CICI framework was also used in the qualitative systematic review on contextual barriers and
facilitators to the implementation of home-based palliative care in Europe. Furthermore, the CICI
framework underpinned the extended logic model to assist decision making and provided the framework
for the rapid applicability assessment of rHBPC. .
The qualitative review This successfully incorporated the CICI framework within the methodology of
‘Best Fit Framework Synthesis’ and the findings offered insights into the substructure of the framework
and the contents of the single domains as well as providing good insight into the interconnectedness of
domains within the framework.
Stakeholder involvement Lay and professional palliative care stakeholders have been consulted
throughout the case study in a number of ways as detailed in chapter 3. The added value of stakeholder
involvement throughout this ‘demonstration HTA’ has been illustrated as follows. Stakeholder
consultation in seven countries at the outset of the project identified a number of shared issues of
importance to palliative care stakeholders. These shared issues, along with findings from a review of
review level evidence, helped the research team to identify a focus for the case study. Stakeholders have
validated some of the findings presented (e.g. patient preferences, moderators of treatment effect) and
provided colloquial (informal) evidence based on their experiential knowledge to inform gaps in the
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evidence (e.g. effectiveness). Stakeholders have provided new or additional perspectives on the
evidence, enabling further exploration and discussion of the results and implications of these for practice
and further research. The colloquial evidence from stakeholders has supplemented other types of
evidence collection within the project and can enhance understanding of the rHBPC and HBPC and their
application.
As the above text indicates, stakeholder involvement has been a key part of the case study. Stakeholder
feedback has been positive, highlighting not only the benefits to the project but also to themselves (e.g.
see appendices 34 and 37).
Key conclusions about reinforced and non-reinforced home based palliative care
Reinforced home-based palliative care interventions were neither significantly better nor worse than non-
reinforced home-based care with respect to patient outcomes and most showed no effect across
caregiver outcomes. It is possible that the outcome measures used are not sensitive enough to detect
relevant effects in this population. Professional stakeholders agreed that support for the lay caregiver is
important. They also agreed that there is still room for much improvement in the development,
implementation and evaluation of reinforced and non-reinforced home-based palliative care interventions.
In the absence of cost effectiveness data, expert elicitations provided insight into costs of reinforced
home based palliative care. Cost data supports the implementation of home based models of palliative
care with the potential to be cost saving, largely because of an expected reduced need for hospitalization
of the patient who is being cared for by the recipient of rHBPC. Although most patients prefer to die in the
security of their own home, determining the acceptability, meaningfulness and appropriateness of rHBPC
and HBPC proved challenging in light of the evidence base and challenges associated with HBPC.
Although most patients prefer quality rather than quantity of life and to die at home, evidence suggests
that single patients who live alone are less likely to die at home. Similarly, patients without caregivers;
with uncontrollable physical symptoms; where physicians may not be available or where concerns exist
about responding to sudden changes are also less likely to die at home. Challenges are associated with
home based palliative care in terms of acceptability for informal caregivers too as this may include
adapting to changing roles and relationships amongst the dyad and professionals; preserving patient and
informal caregiver autonomy and avoiding caregiver burden. In terms of meaningfulness, challenges
relate to the alleviating carer burden and subsequent ill health. Additional concerns exist regarding the
availability, accessibility and equity of palliative care provision and the challenges this poses to caregiver
autonomy. A number of barriers and facilitators exist to the implementation of home based palliative care,
notably in supporting informal caregivers in their role. These barriers and enablers related to the provider,
the organisation and structure in which they are embedded as well as the micro-context of the family and
home in which a palliative care patient and his family are living being of particular relevance to the
successful delivery of palliative care. Differences in palliative care provision across Europe means that
careful consideration is required in order to determine which, if any, of the findings could be potentially
applicable across Europe.
Strengths of the case study
Home care is a timely topic of relevance to stakeholders.
The case study reveals the complexity of designing intervention studies in palliative care contexts.
This case study has enabled the application and demonstration of some of the many concepts
and methods developed within the INTEGRATE-HTA project based on what was feasible in terms
of the time and manpower available. To the best of our knowledge, this is the first project that has
developed and applied a ‘process’ model to integrate HTA methods. The INTEGRATE-HTA
model facilitated the integration of very diverse evidence to inform decision making. The value of
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the CICI framework has been demonstrated both within the logic model and the qualitative review
of enablers and barriers to implementation of home-based palliative care services in Europe. A
logic model has been developed to illustrate key aspects of reinforced and non- reinforced models
of palliative care in very complex field.
Additionally, demonstration of a range of key concepts and methods for effectiveness, economic,
ethical, legal, socio-cultural aspects in HTA, along with search strategies to identify evidence for
patient preferences and moderators of treatment effect are all strengths of the INTEGRATE-HTA
project. This application has the potential to influence future HTA methodology.
The inclusion of extensive stakeholder consultations across seven countries is a major strength of
the project as this allowed us to focus on a topic that is potentially of relevance in a number of
countries. Furthermore, the continued involvement of both lay and professional stakeholders
throughout the project has indicated innovative ways in which they can contribute throughout the
HTA process. For example their involvement in providing information for economic modelling, gap
analysis within the effectiveness review and considering the findings of the assessment of patient
preferences and moderators of treatment effect. This is innovative as the involvement of wider
stakeholders largely happens after the HTA is completed but before the decision is made. The
additional involvement of a group of decision makers in a mock decision making meeting was
extremely valuable. The decision making group confirmed that some decision makers would like
to consider more than cost and clinical effectiveness evidence, although this was often not readily
available to them. The weighting exercise indicated the priorities of the decision makers involved
and the discussion assisted in the revision of the final steps of the NTEGRATE-HTA model.
Furthermore, the case study indicates that a variety of methods can be used to engage
stakeholders, with all seemingly being acceptable and effective in terms of providing stakeholders
with a valuable voice within HTA.
The presentation of new (e.g. updated effectiveness reviews for home based palliative care) and
novel (e.g. ‘gap analysis following the effectiveness review) evidence about reinforced and non-
reinforced models of palliative care in a comprehensive manner may direct future research activity
and assist decision making about commissioning future palliative care services.
Limitations of the case study
The case study was hampered by not being associated with a “real world” decision making
process and so the assessment criteria and integration process was not made explicit at an early
stage, making integration more difficult after the identification and synthesis of evidence.
Resource constraints in terms of time and manpower to develop new methods and complete a
standard HTA influenced a) the choice of methods used in this ‘demonstration HTA’ (e.g.
completing a review of reviews rather than a systematic review of primary studies) b) the evidence
that was available to present to the decision making committee and c) the degree to which the
methods have been tested (e.g. limiting the number of steps that were tested in some of the
methods applied).
Reinforced models are underpinned by the COPE theory but it is important to acknowledge that
other theoretical models of caregiving exist and may underpin other models of caregiver support.
Furthermore, we did not develop a typology of interventions targeted at patients and / or carers.
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Limited quantity and quality of evidence exists about reinforced and non-reinforced models of
home based palliative care and the lack of directly relevant evidence in some aspects of the
assessment hampered the case study. As a result, the case study findings are heavily reliant on
information provided by a limited number of experts who tended to describe situations that involve
a single, co-habiting caregiver rather than situations that involve a complex caregiving network.
Little attention was given to dysfunctional family situations where caregivers are not supportive
and homes are not well equipped for caregiving.
The lack of common and widely understood terms for palliative care and the interventions of
interest created major difficulties in achieving a shared understanding among the research team
and with the diverse international range of stakeholders. The terms ‘reinforced’ and ‘non-
reinforced’ home based palliative care are not widely used or recognised in practice, which
coupled with poor descriptions of the heterogeneous models in the literature perhaps resulting in
some lack of clarity about the intervention and comparator for stakeholders.
In practice, similarities and many overlaps exist between reinforced and non-reinforced home-
based models of palliative care which only differ in that reinforced models provide explicit support
to the caregiver. This, coupled with poor descriptions of the heterogeneous models in the
literature, resulted in some lack of clarity about the intervention and comparator for stakeholders.
Although a wide range of stakeholders have been involved in the project, there could have been
greater heterogeneity in the lay and professional groups involved in some consultations (e.g.
socio-cultural stakeholder consultations).
Key areas for further research
Methodological developments for complex health technologies: Further work is needed to test the
guidance developed in the INTEGRATE-HTA project with other complex technologies and develop
evaluation criteria for their success in terms of producing patient centred, integrated evidence.
Stakeholder involvement: Developing and executing a research agenda that closely matches the
questions and concerns of stakeholders remains a challenge. Although there is an increasing expectation
that stakeholders will be involved in project scoping, further work is needed to embed stakeholder
involvement throughout the HTA process. For example, emphasising the potential role that stakeholders
could have in considering HTA findings or contributing to gap analysis. Although robust methodological
approaches and evaluation criteria exist when stakeholder involvement is undertaken as part of
qualitative research, further work is required in this area when stakeholders are involved as advisors
using consultative approaches.
Home-based palliative care: Reinforced and non-reinforced home-based palliative care are good examples of
complex health technologies as they provide many of the conceptual and methodological challenges
typically associated with the assessment of these types of interventions. However, limited evidence exists
to determine whether reinforced models are more acceptable, feasible, appropriate, meaningful, effective,
cost-effective and patient-centred models than non-reinforced models of home based palliative care for
adults. Given stakeholders’ interest in models of home-based care across Europe, it is likely that the
provision of home based palliative care will increase. However, further consideration of the informal
caregiver’s needs and interventions that could be implemented to support them throughout the caregiving
trajectory is required. Having identified some critical questions about how complex palliative care
interventions may be organised and delivered, there is a need to develop specific and carefully focused
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research questions in collaboration with stakeholders before identifying what research methods (both
quantitative and qualitative) are needed to address them whilst taking account of the recognised
complexities.
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Recommendations
Recommendations for HTA
1. Integration from the very beginning of the HTA process:
HTA researchers should frame the research question formatively at the outset of the HTA, and
develop this in light of an understanding of the healthcare system which is the very first step for an
integrated HTA.
In particular, HTA researchers need to consider how the assessment criteria of the HTA research
question will be informed by the evidence collected the ethical, socio-cultural and legal aspects of
the HTA from the outset of the integration process.
The different assessment aspects should be sequentially assessed to address overlaps between
the assessment results (e.g. assessment results regarding patient characteristics can feed into
the assessment of effectiveness).
Further work is needed to ensure that well before specific elements of the evidence required for
the HTA are identified, consideration has been given to the integration process.
Stakeholder consultation should be undertaken at an early stage in the HTA process to elicit an
understanding of the most important criteria for decision making within each context and for each
HTA question posed.
2. Value of concepts and methods:
The concepts and methods developed in the INTEGRATE-HTA project and demonstrated in this
case study should be tested with other complex technologies. This is essential to demonstrate
their value in an HTA undertaken in a “real world” rather than a methodological research project
setting.
3. Complexity:
The complexity characteristics of the intervention should be assessed and taken into
consideration throughout HTA, as they may not be apparent at the outset and their relevance and
significance may emerge during the evidence synthesis process.
4. Stakeholder involvement:
Stakeholder involvement throughout the HTA process should be planned at the outset of the
project and consideration should be given to the positive, negative, short and long term impact of
their involvement in terms of reach and significance. This includes careful consideration of the
range and diversity of different stakeholders who may usefully inform the HTA process and the
interpretation of the findings.
Lay and professional stakeholder at the outset provides valuable insight and understanding of the
complex technology, directing the researcher’s attention to key issues which potentially saves
time.
Stakeholder contributions should be adequately resourced in terms manpower, time and finance
to ensure realistic expectations of what these can achieve.
The purpose and potential impact of the SAPs should be clearly articulated to all stakeholders so
that everyone involved has realistic and shared expectations about such involvement.
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A dialogue should be established with stakeholders throughout the project. Feedback should be
given to stakeholders about the value of their input into the HTA process and the changes made
as a result of the advice or information they provide. An explanation should be given to
stakeholders for decisions made, particularly if this conflicts with the advice and information they
provide.
The advice provided by stakeholders should be viewed as colloquial (informal) evidence based on
experiential knowledge. Experiential knowledge and colloquial should be recognised within the
evidence hierarchy and methods developed to evaluate this.
5. Communication with stakeholders:
The purpose and potential impact of the SAPs should be clearly articulated to all stakeholders so
that everyone involved has realistic and shared expectations
HTA researchers should establish a dialogue with stakeholders throughout the project. Feedback
should be given to stakeholders about the value of their input into the HTA process and the
changes made as a result of the advice or information they provide. An explanation should be
given to stakeholders for decisions made, particularly if this conflicts with the advice and
information they provide. The advice provided by stakeholders should be viewed as colloquial
(informal) evidence based on experiential knowledge.
6. Definitions
Researchers need to invest time and effort in developing a shared understanding of terminology
used within the HTA of a complex intervention. Within palliative care there are a number of terms
which may have different meaning in different settings, or countries, and so developing a shared
understanding of terms is essential amongst stakeholders locally, nationally and internationally.
7. Shared understanding of health care systems
Researchers need to invest time and effort in developing a shared understanding of the health
and social care systems within which complex interventions operate as the system may influence
adoption/applicability of the intervention in new settings.
Developing an understanding of important factors within the health and social care systems and how we
expect interventions to work is essential in the HTA of complex interventions.
Recommendations for palliative care
1. Decision makers need to further consider how best to support informal carers.
Given the lack of both quantity and quality of evidence about all aspects of reinforced and non-
reinforced home based palliative care, there is a need to further consider how best to support
informal carers. Supporting family carers to provide care at home is crucial to enabling patients to
die at home.
2. In addition to cost and clinical effectiveness evidence, decision makers should take account of other
types of evidence that may assist in capturing the complexity of palliative care.
Decision makers indicated their desire to take account of a wider range of evidence. Doing so
relies on HTA researchers’ ability to undertake assessments of a variety of aspects.
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3. Decision makers, Service providers, practitioners, informal carers and patients require a greater
understanding of the potential barriers and facilitators of home based palliative care and strategies
need to be developed to overcome the barriers (e.g. equity of access, providing information and
assisting informal carers to navigate the health care system).
4. Practitioners should assess each individual’s willingness (voluntariness) to take on the informal
caregiving role and their needs for support before they commence in role.
5. Practitioners, informal carers and patients should be alert to the effects of home based palliative care
in terms of changing roles and relationships , the possible stigma and loss of social status
experienced and burden that this may impose on the lay carer.
6. Professionals should be alert to carer’s needs for support and consider ways that their needs can be
met.
7. Interventions to support carers need to be considered within wider initiatives to provide coordinated
palliative care, with mechanism to review and adapt the design of the reinforcement components.
8. Better reporting of the components of reinforced and non-reinforced models of palliative care is
required in primary studies and for review level evidence.
With some adaption of the tool, this could be achieved using the TiDiER framework (14) in
primary research studies as well as reviews. This will enable decision makers to determine the
resource implications associated with replicating models.
Key Messages for palliative care
A number of key messages were identified for service commissioners, health and social care
practitioners, academics, carers and service users in the case study. The messages are of interest to all
stakeholders, although some may have more relevance to commissioners, practitioners, academics or
service users and carers.
1. European populations are ageing; people are living with more complex, chronic conditions and a
greater number of co-morbidities. Many people with chronic conditions have palliative care needs.
2. Palliative care aims to relieve suffering and improve quality of life for both patients and families
throughout a life limiting illness, not just at the end of life. Palliative care is highly complex as it is
interdisciplinary and holistic in nature (i.e. addresses physical. psychological, social, spiritual and
emotional needs) and treats the patient and family as the ‘unit of care’. Palliative care also
provides support for family carers post bereavement.
3. Palliative care is delivered by two distinct categories of health and social care professionals:
specialist and generalist palliative care providers. ‘Generalist palliative care’ providers are health
professionals who have not received specific training in palliative care, but routinely provide health
care for patients at the end of their lives, which may include general practitioners, community
nurses, nursing home staff and hospital staff. ‘Specialist palliative care’ is provided by teams of
multidisciplinary palliative care professionals who have undertaken specialist training in palliative
care and work only within the field of palliative care.
4. Informal carers have a key role in supporting people with palliative care needs, including those
approaching their end of life. Informal carers are lay people in a close supportive role who
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undertake vital care work and emotional management, although they may or may not be family
members.
Care is often provided by a complex network of family, friends and neighbours, each contributing
different aspects of practical and emotional care, and economic resources. Not all families may be
functional or supportive and not all homes may be safe, comfortable or well equipped for end of
life care. Much evidence shows patients have a preference for care and death at home.
Interventions should, therefore, reflect this preference by aiming to deliver care outside
institutional settings where possible to enable patients to remain at home if desired. Care at home
can be rewarding for those closest to the patient, including the informal caregiver. However, the
role of informal carers differs in each family and is known to change over time. Providing home
care may increase the burden of informal caregivers close to the patient, causing physical,
psychological and social stress both during care and after the death of the patient.
5. This is important as informal carers are known to suffer high levels of stress and burden as a
result of their caregiving responsibilities. Caregiver stress and burden may result in a breakdown
of informal caregiving and increase costs and health service use should the carer become ill
themselves or unable to continue in their role.
6. As a priority, informal caregivers need equitable access to high quality, flexible services that are
able to respond to individual needs and circumstances. There may be a limited window of
opportunity to support informal carers, whose needs may change over time and with the patient’s
condition and their healthcare needs. Additionally, provision of support for informal caregivers
relies on them being identifed and many people do not consider themselves to be ‘carers.’
7. A number of interventions and services have been developed to support informal carers.
However, concerns exist regarding the availability, accessibility and equity of palliative care
provision. Tailored, responsive support may be needed because each family and caregiving
situation is unique.
8. Home based palliative care services have been developed with and without an additional
component of caregiver support. These models are known as “reinforced” (rHBPC) and “non-
reinforced” home based palliative care (HBPC) respectively. Although diverse (heterogenous) in
nature, reinforced (rHBPC) and non-reinforced models of home based palliative care (HBPC) may
be similar in several respects. HBPC models primarily focus on patients, though support may
also be directed towards caregivers on an ad hoc basis rather than as specific planned
intervention. rHBPC will always include an intentional, explicit attempt to support informal
caregivers in addition to the care given to patients. ‘Support’ is broadly defined as any psycho-
educational intervention aimed at providing assistance to caregivers (e.g. individual or group
counselling, education, advice or respite services). Hence, this support may be more active in
nature, aiming to prepare the caregiver for the patient care to come, by teaching the necessary
skills, or it may be reactive in nature, aiming to address psychological health burden of the lay
caregiver resulting from care. Some reinforced models are underpinned by specific theories,
such as stress-coping while others may not be based on theory.
9. The need to support informal carers in their caregiving role is important and widely acknowledged
in policy and service commissioning documents. However, there is a limited amount and quality of
research evidence about reinforced (rHBPC) and non-reinforced home based palliative care
(HBPC) on which to base decisions about which services should be commissioned. Poor
reporting about the components of rHBPC and HBPC hinders comparisons of the interventions,
increasing limitations relating to the volume of available evidence. Additionally, stakeholders’ lack
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of familiarity with the term ‘reinforced’ palliative care resulted in lack of clarity and shared
understandings of rHBPC and HBPC.
10. Service commissioners should consider the use of mixed methods, qualitative research and
action research as a potential way of evaluating services. Randomised controlled trials and other
types of experimental evidence are often not feasible in palliative care and action research
methods are also a useful way to engage with local clinicians, patients and informal carers, who
are key stakeholders, in the process of service development.
11. Reinforced home-based palliative care interventions were neither significantly better nor worse
than non-reinforced home-based care with respect to patient outcomes and most showed no
effect across caregiver outcomes. It is possible that the outcome measures used are not sensitive
enough to detect relevant effects in this population.
12. In the absence of cost effectiveness data, expert elicitations provided insight into costs of
reinforced home based palliative care. Cost data supports the implementation of home based
models of palliative care with the potential to be cost saving, largely because of an expected
reduced need for hospitalization of the patient who is being cared for by the recipient of rHBPC.
13. Although most patients prefer quality rather than quantity of life and to die at home, evidence
suggests that single patients who live alone are less likely to die at home. Similarly, patients
without caregivers; with uncontrollable physical symptoms; where physicians may not be available
or where concerns exist about responding to sudden changes are also less likely to die at home.
14. Although most patients prefer to die in the security of their own home, determining the
acceptability, meaningfulness and appropriateness of rHBPC and HBPC proved challenging in
light of the limited quality and quantity of evidence about models of home based palliative care
and challenges associated with HBPC.
15. Challenges are associated with home based palliative care in terms of acceptability for informal
caregivers as this may include adapting to changing roles and relationships amongst the patient-
carer dyad and professionals; preserving patient and informal caregiver independence in decision
making (autonomy) and avoiding caregiver burden.
16. In terms of meaningfulness, challenges relate to alleviating the carer burden and subsequent ill
health. Additional concerns exist regarding the availability, accessibility and equity of palliative
care provision and the challenges this poses to caregiver autonomy.
17. In terms of feasibility, a number of barriers and facilitators exist to the implementation of home
based palliative care, notably in supporting informal caregivers in their role. These barriers and
enablers related to the provider; the organisation and structure in which they are embedded as
well as the micro-context of the family and home in which a palliative care patient and his family
are living being of particular relevance to the successful delivery of palliative care. For example,
communication difficulties arise from a variety of causes (e.g. patient capacity, physician / staff
paternalism or service restrictions on choices available). Lay carers’ willingness to undertake the
role and be supported in it influences the situation. Some barriers related to informal carers’ lack
of willingness to disclose their needs and other barriers related to professional carers’ inability to
identify caregiver needs, lack of training or resources. The feasibility of HBPC is associated with
the number of agencies involved in providing care and the degree to which services are
coordinated, which may be influenced by professional training, cultures and cooperation styles.
Furthermore, access of the informal caregiver to the health and social system, difficulties in
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navigating the system as well as barriers to actually utilizing them were often reported. All these
issues need to be considered when making decisions about how best to support patients and their
carers.
18. The limited amount and quality of evidence that was available about rHBPC and HBPC and the
lack of directly relevant evidence in some aspects of the assessment impeded the conclusions
that can be drawn from the application of the assessment methods applied in this case study. As
a result, the case study findings are heavily reliant on a limited number of experts. Careful
interpretation is required when considering the case study results.
19. Differences in palliative care provision across Europe means that careful consideration is required
in order to determine which, if any, of the findings could be potentially applicable across Europe.
20. There is little evidence about home based palliative care and it is not clear which services may
best support informal carers, but where possible, support should reflect the needs and
preferences of both patients and carers
21. There is a need for more research to understand how support might best be tailored to meet the
different circumstances and specific needs of different patients and carers.
22. Further evidence is required about all aspects of home based models of palliative care assessed
in this HTA case study. With regard to research examining the effectiveness of home based
models of palliative care, careful consideration should be given to outcome measures to ensure
that these reflect the purpose of the intervention, the choice of research design and patient and
carer perspectives about what is important.
23. Better reporting of the components of reinforced and non-reinforced models of palliative care is
required in primary studies and for review level evidence. This will enable decision makers to
determine the resource implications associated with replicating models.
References
SHARMA, T., CHOUDHURYM., KAUR, B., NAIDOO, B., GARNER, S., LITTLEJOHNS, P.,
STANISZEWSKA, S. (2015). Evidence informed decision making: the use of “Colloquial evidence” at
NICE”. International journal of technology assessment in health care, 1-9.
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Stakeholder Advisory Panel Members in England22
Professionals
From Sheffield Teaching Hospitals, including the Sheffield End of Life Care Clinical Commissioning Group (CGC) and
St Luke’s Hospice.
Adams, Bev
Ashmore, Trudi
Bowling, Jo
Brookes, David (Dr)
Brown, Jayne (Professor)
Chikura, Gertrude
Cromack-Bird, Katie
Dewar, Sue
Fairbrother, Carole
Grande, Gunn (Professor)
Griffiths, Sian
Hargreaves, Peter
Kyeremateng, Sam (Dr)
Larkin, Philip (Professor)
McTague, Laura
McMillan, Susan
Merritt, Penny
Miller, Liz
Oliver, David (Dr)
Owen, Janet
Park, Judith
Payne, Sheila (Professor)
Reeder, Mike (Captain)
Saunby, Debbie
Seamark, David (Dr)
Shipston, Ginny
Statham, Jo
Stuttaford, Joanna
Sullivan, Mike
Theobald, Rebecca (Dr)
Ward, Eleanor
Wee, Bee (Dr)
Williams, Clare
Wilson, Fiona
Yokney, Kathryn
Patient Participation Involvement Group Members
Bellamy, Rosemary Redshaw, Heather
Cork, Simon Robertson, Don
Gath, Jacqui Sharpe, Ivy
Hall, Ken Thompson, Don
Koroma, Pearl White, John
Lowndes-Smith, Viv Wood, George
Consumer Research Panel Members
Ardron, David Gethin, Alison
Ardron, Judith Green, Tracy
Ashton, James Jones, Frank
Bevan, Julie McDade, Dallas
Bower, Peter Revell, Deirder
Chatterjee, Debjani (Dr) Rouse, John
Gath, Jacqui
22
Please note, stakeholders from Germay, Italy, The Netherlands, Norway, Poland and Lithuania were also involved in meetings throughout the project. Although we do not have permission to name the individuals who have generously given their time and expertise to the project, their input is very much appreciated.
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Researchers
Ryan, Tony (Dr)
Wilson, Fiona