psychosocial aspects of living with congenital heart...

Umeå University Medical Dissertations, New Series No 1532

Psychosocial aspects of living with congenital heart disease child, family, and professional perspectives

Anna-Lena Birkeland

Department of Clinical Sciences

Pediatrics

Child and Adolescent Psychiatry

Umeå University

901 87 Umeå

Umeå 2012

Responsible publisher under Swedish law: the Dean of the Medical Faculty

This work is protected by the Swedish Copyright Legislation (Act 1960:729)

© Anna-Lena Birkeland

ISBN: 978-91-7459-511-6

ISSN: 0346-6612

Cover picture: Annica Anca Elmberg, www.källbergelmberg.se

E-version available at http://umu.diva-portal.org/

Printed by: Print & Media

Umeå, Sweden 2012

Alla är en berättelse… som vi får lyssna till…

PROLOG

Mitt professionella fokus har varit psykosocial utredning, bedömning

och behandling av barn med hjärtfel och familjen runt dem. Jag har

fått ta del av och lyssnat på många berättelser från hjärtbarn, föräldrar

och familjer, var och en av dessa är i sig själva en studie av komplexitet,

anpassning, bemötande och samarbete. Detta kliniska arbete inom

barnhjärtsjukvård har gett mig kunskap om hjärtbarnens livsvillkor,

familjen, professionella utmaningar inom barnhjärtsjukvård samt om

sjukvårdens och samhällets förmåga och ibland, trots goda avsikter,

oförmåga att se och möta behovet av ett helhetsomhändertagande och

upprätthålla beredskap att möta komplexiteten på både individ,

grupp/familj och samhällsnivå. Genom arbetsprocessen med

avhandlingen har jag fått möjlighet att fördjupa och vidare utforska

några av de aspekter som jag identifierat i mitt kliniska arbete.

i

TABLE OF CONTENTS

Table of contents _________________________________________________ i

Abstract ________________________________________________________ iii

Populärvetenskaplig sammanfattning _______________________________ v

Original papers __________________________________________________ ix

Abbreviations ___________________________________________________ x

Thesis at a glance ________________________________________________ xi

Introduction _____________________________________________________ 1 Theoretical framework ________________________________________________ 2

Quality of life _____________________________________________________ 2 Stress - Coping____________________________________________________ 5 Psychosocial approach _____________________________________________ 6 Support _________________________________________________________ 7 Profession _______________________________________________________ 8 Team ___________________________________________________________ 9 Transition _______________________________________________________ 10

Methodological framework ________________________________________ 11 Quantitative methods _________________________________________________ 11

Structured interview: Study I ________________________________________ 11 Questionnaire: Study IV ____________________________________________ 12

Qualitative method ___________________________________________________ 12 Studies II–IV______________________________________________________ 12 Study II _________________________________________________________ 12 Study III _________________________________________________________ 13 Study IV _________________________________________________________ 13 Statistical analysis: Studies I and IV ___________________________________ 13

Ethics __________________________________________________________ 14

Aims of the study ________________________________________________ 15 Overview of the research process________________________________________ 16 Study groups ________________________________________________________ 16

i i

Results ________________________________________________________ 20 Study I: Complexity __________________________________________________ 20

Aim ___________________________________________________________ 20 Design and results ________________________________________________ 20

Study II: Coping ______________________________________________________ 21 Aim ____________________________________________________________ 21 Design and results _________________________________________________ 21

Study III: Profession ___________________________________________________ 22 Aim ____________________________________________________________ 22 Design and results _________________________________________________23

Study IV: Teams/PCTs _________________________________________________ 24 Aim ___________________________________________________________ 24 Design and results ________________________________________________ 25

Discussion _____________________________________________________ 26 Complexity _________________________________________________________ 26

Personal sphere __________________________________________________ 27 Interpersonal sphere ______________________________________________ 27 External sphere ___________________________________________________ 27

Coping – Stress _____________________________________________________ 28 Profession _________________________________________________________ 30 Teams/PCTs _________________________________________________________32

Methodological considerations and limitations _______________________ 34 Quantitative method _________________________________________________ 34 Qualitative method __________________________________________________ 35

General summary _______________________________________________ 37

Challenges for the future _________________________________________ 38

Acknowledgements _____________________________________________ 39

References ______________________________________________________41

i i i

ABSTRACT

Background

The vast majority of infants born with congenital heart disease (CHD) reach

adulthood because of the developments in cardiology in recent decades. This

thesis aims to describe the psychosocial situation of child/adolescent cardiac

patients and their families, investigate the situation faced by parents and siblings

initially and over time, investigate the approaches paediatric cardiologists use in

encountering the family, and describe the teamwork occurring in paediatric

cardiology teams (PCTs) in Sweden.

Theoretical framework: The theoretical framework was based on a quality of life

model applied to children, a stress-coping model, and a psychosocial approach

including support, profession, and teamwork.

Methods

The research combines quantitative data collection/analysis and qualitative

research interviews/content analysis.

Results

Complexity: The three grades of medical complexity differed regarding the

number and severity of psychosocial symptoms, the children with the most

complex CHD having the most severe symptoms. The most frequent symptoms in

the whole patient group regarding various spheres were: healthcare and

treatment-related needs in the external sphere, family symptoms in the

interpersonal sphere, and mental/psychosomatic symptoms in the personal

sphere.

Coping: Being informed of a child’s/sibling’s heart disease has emotional

consequences, so information, communication, and support are essential.

Breaking the news of a child’s disease can be described as a turning point still

significant after ten years. The professionalism of the doctor’s approach in

breaking the news is crucial.

Profession: Among paediatric cardiologists, how to break bad news to a family is

an important concern, evident in findings regarding the significance of trust and

confidence and the use of various emotional positions. Paediatric cardiologists

commonly wish to be skilled at handling this situation, and attaining the needed

skills calls for reflection, education, and sharing experience.

i v

Team: PCTs in Sweden aim and try to work in a structured way. In PCTs, there is

a need for leadership, resource coordination, coaching, and a forum for joint

reflection. Dependence on the physician on the team was identified in all PCTs.

The challenge of managing increasing complexity at both the family and system

levels requires interprofessional teams.

Conclusions

These studies illustrate the psychosocial complexity and the need of psychosocial

support. Emotional consequences, communication, information and support are

essential both for the children, parents/families and for the professionals. To

manage this complexity organizational alteration action plans are required. There

is a need for a forum to stimulate dialogue and common reflection in the local

PCT and at the regional and national centres.

Key words breaking bad news, children/adolescents, congenital heart disease,

communication, content analyse, coping, counselling, interprofessional team,

medical education, patient relationships, paediatric cardiology, professional role,

psychosocial inventory method, qualitative method, quantitative method stress,

teamwork

v

POPULÄRVETENSKAPLIG SAMMANFATTNING

Bakgrund

Sedan flera decennier har barnhjärtsjukvård genomgått en omfattande

utveckling. För 50 år sedan ledde obehandlat medfött hjärtfel till barns död i 60–

70% av fallen. Flertalet barn som är födda med hjärtfel når nu tonåren och

vuxenlivet tack vare den utveckling som skett inom barnhjärtkirurgi,

intensiv/nyföddhetsvård och barnkardiologi de senaste 40–50 åren. Idag kan

barn som tidigare bedömdes vara icke behandlingsbara ges en tillfredsställande

korrigering eller lindring. Nya metoder har revolutionerat behandlingen och

vården av dessa barn men också lett till livslång uppföljning för vissa hjärtbarn. I

denna nya grupp av patienter ingår t.ex. barn med s.k. enkammarhjärtan och de

barn/ungdomar som kan behöva genomgå hjärttransplantation. Erfarenhet från

vården av barn med hjärtfel tyder på att hjärtfelet inte alltid är det största

problemet för dessa patienter utan att de lever med frågor som avser livskvalitet

och den psykosociala situationen. Barn som behandlas för medfödda hjärtfel har

ofta en mycket speciell erfarenhet i livet och kan tvingas leva med olika besvär.

Många behöver ha kontakt under lång tid med hälso- och sjukvården.

Tillsammans med ökade investeringar i medicinsk teknik, behandling och vård av

barn med hjärt- sjukdom finns ett behov av att vidareutveckla och vidare utforska

det psykosociala området för dessa patienter.

Avhandlingens syfte: Det övergripande syftet med denna avhandling var att öka

kunskapen om den psykosociala situationen för barn med medfött hjärtfel (CHD)

och hur de professionella inom barnsjukvården möter barn och familjer som

drabbats av hjärtfel.

Avhandlingens specifika syften:

- att beskriva den psykosociala situationen för barnet/ungdomen och familjen

- att undersöka erfarenheter hos föräldrar och syskon vid beskedet om hjärtfel

och hur man hanterat detta över tid

- att undersöka förhållningssätt i mötet mellan barnkardiolog och familjen

- att beskriva de barnkardiologiska teamen (PCT) i Sverige och deras

förhållningssätt till att arbeta med barn med hjärtfel.

Teori Den teoretiska referensramen utgår från en livskvalitetsmodelll för barn, stress

och coping/anpassning samt psykosocialt förhållningssätt där stöd, profession

och team ingår.

v i

Metoder

Kvantitativa data och kvalitativa forskningsintervjuer har kombinerats.

Kvantitativ metod: Utifrån en livskvalitetsmodell har ett instrument tagits fram

för bedömning av den psykosociala situationen för barn med hjärtsjukdom.

Psykosociala symtom har registrerats. Formuläret delades in i sfärerna personell,

interpersonell och extern.

En enkät har skickats ut till samtliga barnhjärtteam i Sverige.

Kvalitativ metod: Kvalitativa forskningsintervjuer har genomförts med föräldrar,

syskon, barnhjärtläkare och barn-hjärtteam. Intervjudata har analyserats med

kvalitativ innehållsanalys där man systematiskt och strukturerat bearbetar

textdata för att se mönster och teman.

Resultat Komplexitet: De tre graderna av medicinsk komplexitet, skilde när det gäller

antal och svårighetsgrad av psykosociala symptom, där barn med mest komplexa

hjärtsjukdomarna hade de mest frekventa och de allvarligaste symptomen. De

vanligaste symptomen i hela patientgruppen i de olika sfärerna var vård och

behandlingsrelaterade behov i den externa sfären, familjesymtom i

interpersonella sfären och psykiska/psykosomatiska symtom i den personliga

sfären. De allvarligaste symptomen hittades i den interpersonella sfären där

familjesymtom utgjorde den mest allvarliga variabeln. Frekvensen av allvarliga

problem i den personliga sfären var 11% i patientgruppen.

Coping: Att få information om ett barns/syskons hjärtsjukdom får känslomässiga

konsekvenser och information, kommunikation samt stöd är viktigt. Beskedet om

barnets sjukdom kan beskrivas som en vändpunkt i familjernas liv som

fortfarande påverkar dem efter tio år. Läkarens professionella förhållningssätt att

ge besked och sättet att kommunicera är centralt.

Profession: Bland barnkardiologer är hur man ger svåra besked till familjen en

viktig angelägenhet. Tydligt i resultaten är användning av olika känslomässiga

positioner och betydelsen av tillit och förtroende mellan läkaren och

barnet/familjen. Det finns en gemensam strävan att uppnå kompetens att hantera

situationen, och det finns ett behov av reflektion, utbildning och utbyte av

erfarenheter.

Team: Barnhjärtteam i Sverige har ambitionen att arbeta på ett strukturerat sätt

ofta med en sjuksköterska som samordnare. Utmaningen att hantera den ökande

komplexiteten på både familj- och systemnivå kräver insatser av olika

professioner. Barnhjärtteamen möter svårigheter i t ex form av tidsbrist och

problem att samla hela teamet. Inom barnhjärtteamen, finns det ett behov av att

utveckla ledarskap, samordning av resurser, coachning och forum för gemensam

reflektion.

v i i

Slutsatser

De fyra studier som ingår i avhandlingen visar sammantaget på komplexiteten för

barn med medfödda hjärtfel, en komplexitet som både barnet, familjen och de

professionella lever med och måste hantera. Att få besked om barns hjärtfel är

stressande och känslomässigt påfrestande men hanterbart över tid.

Barnhjärtläkarens förhållningssätt har betydelse för föräldrarna och dennes sätt

att kommunicera är betydelsefullt. Emotionella konsekvenser, kommunikation,

information och olika former av stöd är väsentligt både för barn,

föräldrar/familjer och för de yrkesverksamma. För att hantera denna komplexitet

krävs stöd, professionalitet, systemförståelse, organisatorisk insikt och

handlingsplaner för att sätta hjärtbarnet i centrum. Det finns ett behov av forum

för att stimulera dialog och gemensam reflektion i de lokala barnhjärtteamen och

på regionala och nationella centra.

Nyckelord barn/ungdomar, coping, kardiologi teamarbete, kommunikation,

medfödd hjärtsjukdom, medicinsk utbildning, psykosocial symtominventering,

hjärtsjukdom, patientrelationer, rådgivning stress, profession

v i i i

i x

ORIGINAL PAPERS

This thesis is based on the following papers, which are referred to in the text by

their Roman numerals.

I. The complexity of the psychosocial situation in children and adolescents

with heart disease. Birkeland A-L, Rydberg A, Hägglöf B.

Acta Paediatrica 2005;94:1495–1501

Reprinted with permission from the publisher

II. Facing bad news: a case study focusing on of families having a child with

congenital heart disease. Birkeland A-L, Hällgren-Graneheim U, Rydberg A,

Hägglöf B, Dahlgren L.

In manuscript

III. Breaking bad news: an interview study of paediatric cardiologists. Birkeland

A-L, Dahlgren L, Hägglöf B, Rydberg A.

Cardiology in the Young 2011;21:286-291

Reprinted with permission from the publisher

IV. Teamwork in Swedish paediatric cardiology: a national exploratory study

examining function and dynamics. Birkeland A-L, Rydberg A, Dahlgren L,

Hägglöf B.

Accepted for publication

x

ABBREVIATIONS

AD/HD Attention-Deficit/Hyperactivity Disorder

CHD Congenital Heart Disease

CBCL Child Behaviour Check List

CAP Child and Adolescent Psychiatry

DSM IV Diagnostic and Statistical Manual of Mental Disorders IV

GARF Global Assessment of Relational Functioning (GARF) Scale

GUCH Grown-Up Congenital Heart disease

HO Habilitation Organization

HRQoL Health-Related Quality of Life

PCT Paediatric Cardiology Team

QoL Quality of Life

SOC Sense of Coherence

SOFAS Social and Occupational Functioning Assessment Scale

WHO World Health Organization

x i

THESIS AT A GLANCE

Aims Material and methods Results Conclusions

Study I.

Complexity

The complexity of

the psychosocial

situation in

children and

adolescents with

heart disease.

Published.

To describe the

psychosocial situation of

children/adolescents

with heart disease and

their families.

n = 9

Children/adolescents with

congenital heart disease

(CHD) were graded into

three categories with

respect to the complexity

of CHD.

Patients were compared

with age- and gender-

matched healthy controls.

A structured interview

model and clinical

evaluation was used to

collect data.

Results were presented as

means, medians, and

percentages. Differences

between groups were

analysed using contingency

analysis (Chi-square test).

The most frequent symptoms

were healthcare- and treatment-

related needs (71/97) in the

external sphere, family symptoms

(68/97) in the interpersonal

sphere, and mental and non-

cardiac-related somatic symptoms

(19/97) in the personal sphere.

Corresponding numbers in the

control group were treatment-

related needs (15/97), family

symptoms (9/97), and somatic

symptoms (25/97). In the patient

group, 50% of the symptoms were

evaluated as mild, 30% moderate,

and 20% severe. The frequency

of severe problems in the

personal sphere was 11% in the

patients and 1% in the controls.

The study illustrated the psychosocial

complexity and the need for

psychosocial support in the study

group.

The three grades of medical

complexity differed regarding the

number and severity of psychosocial

symptoms. Severe personal problems

were experienced in the most complex

group, indicating that the patients have

severe personal problems independent

of family problems.

In line with development of medical

treatment, the results indicated a need

for structured psychosocial

programmes based on the complex

psychosocial situation of children and

adolescents with heart disease and

their families.

Study II.

Coping

Facing bad news

- a case study

focusing on

families having

a child with

congenital heart

disease.

Manuscript

To explore the

experiences of parents

and siblings of children

with CHD during the

diagnostic phase and

how family members

dealt with the situation

over time.

n = 8

Children with CHD

representing three levels of

complexity from a patient

group of 97 children. Family

members were interviewed

on two occasions ten years

apart and data were

collected from eight semi-

structured interviews.

Qualitative content analysis

was used.

Main themes:

- Being informed of the diagnosis

evoked overwhelming feelings

- Ambivalence towards medical

interventions and technology

- Expressed need for professional,

social, and emotional support

- Pathways to new perspectives

- Outlook on life and future

changed over time

Receiving news of a child’s CHD could

be described as a turning point. How

the problems were evaluated and dealt

with varied over time. Despite marked

stress, living with and caring for a child

with heart disease proved

manageable. In both the short and

long terms, well-functioning

communication between the families

and medical personnel was described

as essential.

Study III.

Profession:

physician

Breaking bad

news: an

interview study

of paediatric

cardiologists,

Published.

To examine the

approach of paediatric

cardiologist in informing

and communicating with

the family and patient.

n = 9

Nine semi-structured

interviews were carried out

with paediatric

cardiologists.

Qualitative content analysis

was conducted.

Main themes:

- Method of providing information

- Emotional positions

- Creation of confidence

- Interaction

- Success and failure

- Strategies for handling the

situation

- Various themes/needs all

involved a need for reflection

- Development of teamwork is

required

Physicians were expected to cope with

the complexities of diagnoses and

decisions, while being sensitive to the

feelings of the parents, aware of their

own emotions, and able to keep all this

under control in the context of breaking

the bad news to the parents and

keeping them informed. These

conflicting demands created a need to

expand the professional role by

including more training in emotional

competence and communicative

ability.

Study IV.

Team: PCT

Teamwork in

Swedish

paediatric

cardiology:

a national

exploratory study

examining

function and

dynamics,

Accepted.

To describe PCTs in

Sweden, their

organization and

function, and to deepen

our understanding of

working in a PCT.

n = 30

Questionnaire sent to all 34

contact nurses for

paediatric cardiology in

Sweden.

n = 6

Focus group interviews with

PCTs selected by size and

location. Statistical and

content analysis were used.

Thirty of 34 PCT units completed

the questionnaire. The nurse was

coordinator in 17/26 units.

Main themes:

- Organization/ way of working working

- Function and task

- Attitude/approach

- Communication

The challenge was to meet and

manage the increasing complexity.

Structural/organizational limitations

and doctors’ working conditions were

crucial factors. The PCTs need

leadership, team coordination/

communication, support, continuing

education, and access to professional

management consultants. Fora for

dialogue and joint reflection are

needed in the local PCT and at

regional and international centres.

I n t r o d u c t i o n | 1

INTRODUCTION

Paediatric cardiology has been developing extensively for several decades. Fifty years

ago, children died from untreated congenital heart (CHD) disease in 60–70% of

cases. Today, however, most children born with CHD reach adolescence and

adulthood thanks to developments in cardiac surgery, interventional cardiology,

intensive/neonatal care, and general paediatric cardiology over the past 40–50 years

(1–3). Childhood conditions once deemed untreatable can now be adequately

corrected or alleviated (4). Treatment and care of patients with CHD have been

revolutionized by the introduction of new methods in surgery and anaesthesiology

and the development of specialized children’s intensive care units, though this has led

to life-long follow-up for some children with CHD (5). This new group of patients

includes children with single-ventricle heart defects and children/adolescents who

need to undergo cardiac transplantation (2, 6–10). Although the outcome of

corrective cardiac surgery is the key factor for survival, other factors such as

psychological and social issues are of great importance in terms of quality of life (11).

Clinical experience suggests that the cardiac defect is not always the most serious

problem facing these patients; instead, quality of life and the psychosocial situation

are the major issues in their lives (12). A holistic approach to care, psychosocial

assessment resources and psychosocial treatment, when appropriate, are important

determinants of the quality of life of the child, adolescent, and the family (11, 12).

Research into heart disease in children and adolescents has previously been

predominantly medically oriented; few psychosocial studies have been carried out in

this field, in contrast with the fields of childhood and adolescent experience of more

common diseases such as cancer, asthma, and diabetes.

Children treated for congenital heart defects often have a very special life experience

and may be forced to live with various difficulties and disabilities. Many require long-

term contact with the healthcare system, doctors, nurses, and paediatric cardiology

teams (PCTs) (13). Studies attempting to highlight quality of life in children with

CHD, as well as psychosocial studies, all identify a complex set of psychosocial

problems (14–17). This patient group also occasionally appears to have behavioural

and emotional problems. Various studies (17, 21-25) have found a higher frequency of

psychological problems in the group with complex congenital heart defects.

The situation of children with CHD and their families is often complex, resulting in a

complicated psychosocial existence involving hospitalizations, dependence on the

healthcare system, and stress to the patients, their parents, and families (26–28).

Professionals face constant challenges in meeting the needs of these children and

their families, helping them deal with the complexity of this situation. As the number

of patients who survive grows, so to does the need for both medical and psychological

care (17). At the same time that paediatric cardiac care is evolving, socioeconomic

changes are also occurring, for example, in the social insurance system. Such changes

2 | A n n a - L e n a B i r k e l a n d

affect the already vulnerable paediatric cardiac patients and their families (29, 30).

The lack of holistic care may therefore also have economic consequences for these

children and their families/parents, with the risk of worsening financial conditions

(31).

Along with increased investments in medical technology, treatment, and care for

children with CHD, the psychosocial situation of these patients needs further

development and exploration (17, 23, 32). Studies from a psychosocial perspective

with a focus on quality of life that highlight the need for further research in this area

reveal a psychosocial perspective in paediatric cardiac care, thereby contributing to

improved quality of life for children with heart disease and their families. Bio-

psychosocial models that integrate a biological perspective have increasingly been

advocated for evaluating the effects of healthcare. The World Health Organization

(WHO) has been involved in the development of such a system addressing quality of

life, namely, the ICF-C4 (33).

Theoretical framework

The theoretical points of departure are quality of life (QoL) (34), stress – coping (35,

36), and psychosocial approach (37) including support, profession, and teamwork.

Quality of life

The aim here was to describe the complexity of the situation and contribute to an

overall picture of the psychosocial situation of children with CHD. The QoL concept is

relevant because its definition is multifaceted, representing a complex composition of

various aspects of human life and of the individual’s perception of his/her life

situation – i.e., the culture and value systems in which people live as related to their

goals, expectations, benefits, and concerns (34). The QoL concept is integral to many

scientific disciplines and is interdisciplinary in nature, which enables and creates

opportunities for exchanging knowledge between disciplines and professions, such as

philosophy, sociology, economics, psychology, and medicine. This is a good starting

point from which to describe and understand the complexity of the psychosocial

situation of children with CHD.

Lindstrom presents a relevant theoretical summary of the QoL concept in The

Essence of Existence (34); the model used in the present thesis is based on parts of

Lindström’s theoretical model. In healthcare research, the conceptual content has

shifted towards the health-related quality of life (HRQoL) concept (38), which is a

multidimensional concept that includes domain related to physical, wellbeing and

social functioning. It goes further than direct measures of population health, and

focuses on the impact health status has on quality of life (38).


Various instruments and questionnaires have been developed to measure the child’s

HRQoL. Some instruments are designed to assess the effects of the treatment of a

disease, while others analyse how a particular disease affects QoL.

Examples of instruments for measuring QoL in children with chronic diseases

include PedsQoL (Paediatric Quality of Life Inventory) (39), DisabKids (40), and

Kidscreen (41). PedsQoL includes a specific cardiac module (42), and another

cardiac-specific HRQoL instrument is the Paediatric Cardiac Quality of Life

Inventory (PCQLI) (43). At the time that we planned this study, however, no such

instruments were normed for use in Sweden. We also hoped to capture QoL from a

broader perspective in order to characterize the complexity associated with children

with cardiac disease using a method that would allow us to measure and describe

their psychosocial situation in general. Moreover, the age range of the studied

children was large, and it was difficult to find an instrument that covered all ages.

In The essence of existence, Lindström describes a QoL model in which he divides life

into four spheres: personal, interpersonal, external, and global (34) (Table 1).

Table 1. Quality of life model for children (Lindström).

Spheres Dimensions As related to children

Global Ecological, social, and political resources

1. Macro environment 2. Culture 3. Human rights 4. Welfare policy

Physical environment, attitudes, compliance with the Children’s Convention, distribution of welfare

External Social and economic resources

1. Work 2. Income 3. Home

Parents’ education, work and satisfaction; distribution of income and satisfaction; housing, space, and room for child

Interpersonal Access to social relationships and support

1. Family structure and function 2. Close friends 3. Expanded social support

Number of siblings, number of adults in household, satisfaction with family; support from friends, neighbours, and community

Personal Personal resources

1. Physical 2. Psychological 3. Spiritual

Growth, activity, self-esteem, basic mood, meaning in life

In consultation with Lindström, we chose to illustrate the personal, interpersonal,

and external spheres using circles in which we place the child at the centre

surrounded by interactive factors. Consequently, our modified model does not

include the global sphere (Fig. 1).


Personal

Interpersonal

External

Figure 1. Quality of life model for children.

According to Lindström, for life to be rich in quality, key components must be

included in the QoL concept and these areas of life must be important to the

individual. According to Table 1, Lindstrom’s model is covered by resources

distributed between four spheres/systems: personal/individual resources, family and

other interpersonal resources, social and economic resources, and social and political

resources. According to the model, families define QoL and what improves it. If

family members receive help with the problems for which they feel they need help, a

serious illness or handicap does not have to reduce their QoL.

In recent years, research has focused on understanding and describing QoL. We can

follow developments in this field in a review by Lindström and Eriksson (38). A key

researcher in this field is Antonovsky, who contributed to our understanding of

salutogenics (44). Antonovsky’s research focused on what causes people to become

and remain healthy, i.e., the origin of those factors that are salubrious or salutogenic.

The concept of sense of coherence (SOC) was Antonovsky’s answer to the question of

what renders life salutogenic (44, 45). According to Antonovsky (45), SOC is a global

orientation expressing a pervasive, enduring, and dynamic feeling of confidence that

the internal and external worlds are predictable, and that there is a high probability

that things will go as well as can reasonably be expected. SOC can be divided into

three components: comprehensibility, manageability, and meaningfulness. An


individual with a high SOC understands what is happening in a given situation, can

manage the situation, and sees meaning in what is happening, while an individual

with a low SOC perceives the situation as incomprehensible, unmanageable, and

meaningless. According to Antonovsky, comprehensibility refers to how a person

experiences internal and external stimuli, either in the form of order, clarity,

structure, and coherence, or as incoherent and inexplicable chaos and ambiguity.

Manageability refers to the capacity of an individual to cope with problems, using the

available personal or external resources. Meaningfulness is the third component of

SOC, and people with a high sense of meaningfulness feel it is worth dedicating

energy and commitment to solving problems. They view life with confidence and

believe that life has purpose and meaning (45, 46). The strength of Antonovsky’s

theory of SOC is that it focuses on a person’s entire “lifeworld” and how events and

circumstances shape how the individual sees him- or herself. Antonovsky believes it

is a person’s overall view of life and not one-time events that affect health. According

to Antonovsky, it is mainly the environment surrounding the individual that affects

health, and a low SOC can be changed with the right interventions. Converted to the

QoL concept, this means that, regardless of disease or other stress, a high sense of

context can contribute to a higher QoL.

Stress - Coping

Key life events such as divorce, grief, unemployment, illness, the addition of a new

family member, or the illness of a child may be consistent with the definition of

stressors and events to which the individual has no automatic response.

Karasek uses the demand–control model to describe how psychological and physical

stressors in life and the decision latitude of the individual affect health (47).

According to Karasek, experiences of stress are related to how well an individual

responds to two protective psychological mechanisms: the “alarm reaction” and

“adaptation”. The “alarm reaction” is the physical reaction that occurs when we are

confronted with dangers, such as information about the serious illness of a child.

“Adaptation” or coping means that we learn what stimuli in the environment no

longer pose a threat to us and adapt to this knowledge. It is crucial that people adapt

to such conditions, otherwise the risk of both psychological and physical collapse is

fairly great. When one of the “alarm reaction” or “adaptation” mechanisms does not

work, what we call stress arises. A situation characterized by high demands combined

with weak ability to control risk leads to stress and “strain”, a situation that the

individual and, in our case, the family wants to avoid. Karasek and Theorell (48)

divide the psychological demands of the sort experienced by the studied families into

two types: cognitive and emotional. People have to cope with situations they are

unprepared to cope with either cognitively or emotionally. The control in their model

also has two facets, involving the personal ability to cope with the situation that

arises, and the positive opportunity for growth. Even extremely pronounced negative

stress of the type addressed here can generate positive growth. Another measure of


negative stress is whether the individual personally experiences the conditions as

stressful and psychologically demanding.

Coping is defined as constantly changing cognitive and behavioural efforts to manage

specific external and/or internal demands that are appraised as taxing (49) or

exceeding one’s resources (50). A range of coping behaviours has been identified.

Lazarus et al. distinguish between problem-focused and emotion-focused coping:

problem-focused coping involves confronting a problem to reduce the effect of a

stressor, while emotion-focused coping involves dealing with the emotional distress

occasioned by the stressor.

A key element of the adaptation concept is that of successful coping. When an

individual and a family has encountered adversity and coped well with the challenge

of, for example, having a child with heart disease, they have used various individual,

family-related, or societal coping resources. All these capacities are defined as factors

that can promote good outcomes despite adversity. The capacity to overcome,

endure, or handle life problems in a productive way can also be described as

resilience, which also implies that individuals can develop and be strengthened by

adversity. Walsh et al. (51) define resilience “as the capacity to rebound from

adversity, strengthened and more resourceful … It is an active process of endurance,

self-righting, and growth in response to crisis and challenge … the ability to

withstand and rebound from disruptive life challenges” (p.4.). The SOC model of

Antonovsky with its holistic view of health fits well within a resilience model. From a

broader perspective, resilience depends on a bio-psychosocial model, as

genetic/hereditary and psychosocial resilient factors can promote good outcomes

even after very negative life experiences (52).

Compas et al. describe the relationships between coping and other reactions to stress

(53). Mackay (54) summarizes these relationships as follows: “Coping is a conscious

intentional response to stress. Coping is often invoked to represent competence and

resilience. However, these three terms have distinct meanings. Where coping refers

to adaptive response to stress, competence refers to the characteristics needed for

successful adaptation, and resilience is reflected in outcomes where competence and

coping” have been displayed (p. 105). In any case, over time, these terms and

concepts have formed the building blocks of our understanding of how individuals,

children, and families manage and adapt to stressful life events and living conditions.

Psychosocial approach

The psychosocial concept is included as part of the theoretical framework in this

thesis, and what follows is an overview of the concept and how it is used. The

Encyclopaedia Britannica and the Swedish National Encyclopedia describe the word

psychosocial as referring to the relationship between social factors and individual

beliefs and behaviour (55). The social conditions of an individual may cause

psychological reactions and vice versa. Factors that can affect the psychosocial


situation of an individual either negatively or positively include grief, joy, love,

illness, finances, work, and the surrounding environment.

Applying a psychosocial approach to clinical practice involves taking account of how

the social and psychological life environments of the individual influence the human

body; for example, physiological effects, behaviours, psychological, and physical

illness.

“Psychosocial” is not a new concept, but can be traced far back in the history of

professional social work (37). In 1941, the social work theoretician Gordon (56), a

representative of the diagnostic school, said: “We must study the whole... Without the

whole, we cannot understand the parts, nor can we understand the whole without the

parts”. She also coined the concept of “the person-in-the situation” with a particular

emphasis on the interaction between the person and the situation. Bernler and

Johnsson (37) define psychosocial work as the social work carried out with

individuals, families, and groups for preventive purposes; they also believe that

studying and understanding the figure/person in question/situation, as described by

Gordon, entail having a psychosocial approach.

The psychosocial approach entails seeing the individual in the context in which

physical, psychological, and social factors interact, having a crucial impact on the

development of a person’s identity and life situation. According to Bernler and

Johnson (37), this approach describes an understanding or attitude in working with

people. Psychosocial work, as described above, takes into account the personal,

interpersonal, and external spheres and, for change to occur, may require

interventions/support in one or more of these spheres.

To describe the practical work based on this approach, we talk about psychosocial

work/treatment, which can be said to be a generic term for the methods within social

work used in relation to individuals, families and groups for preventive or treatment

purposes (57). In the context of this thesis, the psychosocial approach encompasses

various methods for strengthening the resistance of the child/family to external

adversities and making changes in the social environment that would enable the

child/family to better cope with threatening situations (27).

Support

One resource in this effort to cope with stress and strain has been identified as “social

support”, a positive factor that is expected to have a dampening effect on perceived

stress (37). Many providers of medical, nursing, and psychosocial services state that

they use social support as a method. Epidemiological research defines social support

as the presence of people in the vicinity of the individual who can help the individual

with both emotional and practical problems. Bernler et al. believe that certain

activities must be taken to be able to call something support (37, 58). They argue that

support should not be considered just a method of work, but as a component of all


social work, from the first session to the last. In this context, support entails creating

a treatment relationship based on trust. Social support can be divided into two

components, i.e., emotional and professional support, so families may receive

support from friends and acquaintances, as well as from doctors and other healthcare

professionals.

The various methods of psychosocial work can be collectively defined as general

targeted interventions or preventive psychosocial work, that entail informing and

educating various types of groups (57, 58), for example, team development,

educational initiatives, and staff supervision. Psychosocial work in paediatric cardiac

care also aims to improve the psychosocial prognosis of the child and family subject

to the limitations imposed by the cardiac disease (57). The profession offers

individual and family-focused interventions including counselling, identification of

psychosocial situations, information, as well as support for the stresses, strains, and

interaction between involved parties.

Profession

A primary purpose of encounters between patients and professionals is to generate

feelings of trust. In today’s society, the various roles involved in the healthcare

professions, such as those of the doctor and nurse, must meet dual demands, i.e., for

professionalism in the sense of effective treatment based on scientifically proven

experience and for emotional skills when meeting with clients (59, 60). The

professionalism of the healthcare approach increasingly tends to include both

cognitions and emotions, and the discussion that the work should benefit the patient

(60). Consequently, the personal emotions and needs of healthcare staff must be held

in check in favour of displaying respect, interest, and warmth and taking a personal

approach (61).

Emotional work as a concept was recognized in 1979 by Hochschild (62). She

demonstrated that emotional work tended to be invisible, something that did not

count, a kind of voluntary work that, on top of ordinary professional tasks, was often

perceived as burdensome. Many professions have been studied from this perspective.

In a 2002 study of the working conditions of nurses, Bones (63) identifies change

processes in healthcare practices that influenced the emotional work of nurses and

how they perceive those changes (63). Bones found that the combination of

“invisible” emotional work and clinical/documentary work was perceived as

extremely stressful, but that some nurses managed to coordinate the two modes and

satisfy the conflicting expectations of their professional role.

In modern society, trust is increasingly contingent and vulnerable, particularly as it

pertains to the role of the doctor, who is highly dependent on real and perceived

professionalism. British sociologist Anthony Giddens describes this in terms of trust,

which has two faces: one assumes an objective and appropriate treatment, while the

other is based more on who a person is and his/her relationship to the professional


party. Giddens argues that modern society requires that people trust each other and

that this trust also applies to strangers. For example, patients have the right to expect

the same treatment from doctors and other healthcare professionals as do any other

people in the same situation. Society is quite simply based on this foundation, and

trust is the glue that creates societal cohesion and effectiveness. Consequently, if

meetings between a professional and a client fail because the professional does not

meet client expectations, trust in the professional will be shaken and, by extension, so

will trust in the entire profession and ultimately in professionals in general.

Something valuable is created in these meetings, which Giddens describes as “access

points”. People assume each other’s roles in these meetings, at best resulting in “joint

ventures” or at worst generating disappointment and distrust (64).

Team

The various professions involved in interprofessional work associated with children

with CHD are confronting a rapid increase in relevant knowledge. Because of this

knowledge proliferation, no single person can have even an overview of all existing

knowledge in the field. This means that case management requires interprofessional

teams consisting of representatives of various professions, who jointly handle the

complexity and associated challenges of the patient’s psychosocial situation (65, 66).

In this regard, paediatric cardiology teams (PCT) meet the needs of patients and their

families, as well as of the professionals involved in their care.

Teamwork can be viewed from various theoretical perspectives. From the

perspectives of organizational theory and efficiency management, teamwork can be

regarded in terms of decision-making, goal attainment, and interpersonal dynamics

(67, 68). A team can also be understood through the lens of group development

models in which the team is perceived as developing in more or less fixed sequential

stages (69). The concept of team is frequently used in healthcare without being

defined, but implicitly refers to a group of people who to some extent coordinate their

actions. How the terms “multidisciplinary”, “interdisciplinary”, and

“interprofessional” are used is complex and problematic, and they are not interpreted

uniformly or coherently in healthcare (70–72). In the multidisciplinary team,

consultations are arranged, but little or no communication occurs between the

various specialists. Interdisciplinary teamwork, while also based on individual

assessment, is followed by meetings in which team members seek consensus (73).

The theoretical framework of interprofessional teamwork, as mentioned above, is

extensive (74). Hall and Weaver (75) and Kvarnström (76) have discussed the

interaction and mutual dependency of team members, and their responsibility for

patient care, as a continuum extending from “multi” through “inter” to “trans”.

Freeth (77) defines teamwork as “the process whereby a group of people, with a

common goal, work together, often, but not necessarily, to increase the efficiency of

the task in hand” (p. 16). Overall, the team’s role is to see itself in a larger context,

1 0 | A n n a - L e n a B i r k e l a n d

reflecting on patient conditions, on team organization and mission, and on meetings

with patients, colleagues, and other professional (78, 79).

Transition

As one objective of this thesis is to elucidate the psychosocial complexity of the

situations of children with CHD and their families, transition in the context of CHD

could have been a theoretical framework (80). Transition is a middle-range theory

that covers the ambition to understand complex situations and represents a concept

that could also be used as a tool to describe and understand specific and practical

phenomena (80). In this thesis, however, we chose a theoretical framework

combining multiple theories that cover the ambition and aim of this thesis.

M e t h o d o l o g i c a l f r a m e w o r k | 1 1

METHODOLOGICAL FRAMEWORK

This thesis aims to build a deeper understanding of what children with heart disease

and their parents experience by highlighting in various ways the psychosocial

situation of children with cardiac disease. By combining qualitative and quantitative

methodologies and approaches, we broadened the database, resulting in a more

reliable basis for interpretation, improving the possibility of describing the target

group in terms of the complexity of heart disease and allowing for a deeper

understanding of this group’s needs (81–85). Specifically, we combined methods

involving both quantitative data collection and interviews. Quantitative methods

were used in studies I and IV, while qualitative methods in the form of qualitative

research interviews/themed individual and group interviews were used in Studies

II-IV.

Quantitative methods

Structured interview: Study I

The QoL model, previously described as part of the theoretical framework (Table 1,

Fig. 1), was operationalized, resulting in an instrument for assessing the psychosocial

situation of children with heart disease. The research group formulated a new

instrument because at the time of the study no standardized cardiac-specific

instrument for children was available and no single existing instrument could supply

the comprehensive information desired. In addition, except for one subject who was a

grown-up CHD (GUCH) patient, the studied children and adolescents were aged 0–

18 years, which made it difficult to find a scale that covered the entire age span.

Accordingly, an interview-based instrument using structured questions about

psychosocial symptoms was developed. The questionnaire was divided into items

covering the personal, interpersonal, and external spheres. The personal sphere

describes symptoms directly related to the child. The interpersonal sphere describes

symptoms related to the child’s immediate circle, i.e., family, friends, and school. The

external sphere describes symptoms related to medical care, social services, and

finances. Psychosocial symptoms were registered using the questionnaire. An

experienced clinical social worker interviewed the children and their parents. The

interviews covered the predetermined symptom areas. When developing the

assessment instrument, various rating scales and assessment instruments were used

to delineate and define issues such as CBCL (86, 87), definitions from DSM-IV (88),

diagnoses of certain diagnostic areas and the GARF relationship assessment (88),

and the SOFAS functional rating scale (88) from DSM-IV (88, 89).

The symptom areas assessed in the children were based on DSM-IV. GARF (88) was

used to describe family symptoms in the interpersonal sphere. Certain themes

regarding the interpersonal functional problems of the children were taken from the


SOFAS scale (Appendices I and II in Study I). Support for the above approach, citing

examples from various instruments, can be found in the Comprehensive Handbook of

Social Work and Social Welfare (90).

Symptom severity was evaluated and scored using a four-point Lickert scale ranging

from 0 to 3, where 0 indicates no symptoms, 1 mild symptoms, 2 moderate

symptoms, and 3 severe symptoms. A manual was formulated to guide the

questionnaire administration and to determine the weighting of the symptoms (Study

I, Appendix II).

Questionnaire: Study IV

This questionnaire comprised an inventory of 19 questions (Study IV, Appendix I).

Initially, background data were requested: respondent profession, size of catchment

area, number of children enrolled, and number of visits per year. Team organization

was covered by six items, followed by a question containing seven variables, relating

to team function and tasks. Under the heading “Way of working” was items about

meeting frequency, memos, documentation, and organized networking events.

Finally, inquiries were made about team members’ attitudes to teamwork, thinking

about teamwork, and views of what characterizes good teamwork.

Qualitative method

Studies II–IV

The method used to analyse the texts/interviews was interview analysis in the form of

qualitative content analysis (91). The actual process can vary, but the following

description according to Granheim and Lundman (92) applies to the process we used

to analyse the interview texts. The entire text (i.e., analysis unit) was first read

through several times to gain a sense of the whole. Particular sentences or phrases

containing information relevant to specific issues were selected; surrounding text was

included to preserve the context of these passages, which constituted meaningful

units. These meaningful units were condensed to shorten the text while retaining all

the content. The condensed meaningful units were coded and grouped into categories

reflecting the core message of the interviews. These categories represent the manifest

content. Finally, themes were formulated to reveal the latent content of the

interviews.

Study II

In this study, which was based on themes from the above-mentioned personal,

interpersonal, and external spheres (Fig. 1, Table 1), the parents and siblings of

children with CHD were interviewed on two occasions ten years apart. The

interviews, which were semi-structured and lasted two to two and a half hours, were

M e t h o d o l o g i c a l f r a m e w o r k | 1 3

all conducted by one of the authors. The interviews sought to trace the path of the

child and family through their experience of the disease and covered the following

topics: course of the disease, medical care, the child with heart disease, perspectives

of family members, and support. Follow-up interviews, also based on the three

spheres, were conducted with siblings and parents after ten years. Data were

analysed using content analysis.

Study III

Nine experienced paediatric cardiologists were interviewed for one and a half to two

and a half hours each. All interviews were carried out by one of the authors. The

interviews were semi-structured, covering five themes and including follow-up

questions and opportunities for the interviewee to add personal reflections – the

interviewed cardiologists were given great latitude to describe their experiences and

provide in-depth information (81). The main themes were the doctor’s self-

presentation, professional attitudes, and encounters with parents as well as the

messages they conveyed to patients and their families. These themes included topics

such as access to evidence-based knowledge, emotional reactions, and the balance

between proximity and distance in client meetings. Two researchers, a medical

sociologist experienced in qualitative data analysis and a paediatric cardiologist, read

and independently analysed the decoded data, identifying the main themes. Data

were analysed using content analysis.

Study IV

Six PCTs selected by size and location were studied via focus group interviews. The

focus group interview was considered a natural environment in which team members

could share their experiences and opinions and be inspired by each other (93). The

interviews, which lasted one and a half to two hours, were all carried out by one of the

authors. The interviews were semi-structured and team members were given great

latitude to describe their experiences and provide in-depth information (81). An

interview guide covered the following topics from the questionnaire: organization

and way of working, function/task, attitude/approach, communication, and

challenges facing the team. All data were subjected to qualitative content analysis (91,

92, 94, 95).

Statistical analysis: Studies I and IV

Results were presented as means, medians, and percentages. Differences between

groups were analysed using contingency analysis (chi-square test, Fisher’s exact test).

A p-value less than 0.05 was considered to be statistically significant. The Statistical

Package for the Social Sciences (SPSS/PC) and Microsoft Excel were used for

statistical analysis of the data.


ETHICS

All methods and the project as a whole were approved by the Research Ethics

Committee of Umeå University on 14 February 2000; additions to the project were

approved by the Regional Ethical Review Board of Umeå on 24 August 2010.

Before each questionnaire survey or interview, respondents were given information

about the purpose of the research, why we were interested in their participation, and

their rights as research participants, such as the right to withdraw from the study at

any time without specifying any reason and without affecting their right or access to

care and treatment. The information was provided orally in connection with visits to

the clinic and in writing by e-mail or regular mail.

While conversations/interviews could have therapeutic value, for example, by

facilitating the processing of past experiences, they could also rekindle past traumas.

The present interview studies were conducted by an interviewer with extensive

clinical experience in counselling; this ensured an approach to people and interview

material that included professional assessment during the session of the need for

follow-up. The ethical implications of the study did not entail any clear risk to the

respondents.

A i m s o f t h e s t u d y | 1 5

AIMS OF THE STUDY

The overall aim of the study was to increase our knowledge of the psychosocial

situation of children with CHD and of the professions handling them.

The specific aims were:

describe the psychosocial situation of the children/adolescents with CHD and

their families

explore the situation faced by parents and siblings initially and over time

investigate the approaches used in encounters between paediatric cardiologists

and the families of affected children

describe PCTs in Sweden and their approaches to their work

The specific aims to be achieved throughout the research process were addressed in

four studies exploring the three inner spheres of the QoL model (34) (Fig. 2).

Interpersonell

Extern

Personell

I

II

III

IV

I

I

II

I ComplexityII CopingIII ProfessionIV Team

Figure 2. Study I deals with the psychosocial complexity in personal, interpersonal and external spheres. In Study II, describing the strategies for mainly the surrounding family, the interpersonal and external spheres were explored. The paediatric cardiologists and the paediatric cardiology team, in the external sphere, are described in Study III and IV.


Overview of the research process

Clinical realities, together with the goals of treating children with CHD holistically

and of developing a more patient-centred approach, shaped the research team efforts

to inventory the psychosocial situation of children with CHD.

The inventory revealed the complex situation of children with CHD and their

families, in the personal, interpersonal, and external spheres, which illustrated and

clarified several levels of needs. The results indicated the complexity of the situation

in which children with heart disease and their families live (Study I). Children with

heart disease, along with their parents and siblings, have to cope with and adapt to

complexity and vulnerability and find a path through these life circumstances.

Naturally, the next step in the research process was to describe the experiences and

coping strategies of these patients and families. The experience of being informed of

the diagnosis and of the needed interventions was described as highly emotional and

still affected the lives of the children and their families 10 years later. Parents stated

that the doctors and the approaches they used were important and that, in particular,

receiving the news about the heart defect was central to their experience (Study II).

Doctors and their profession thus came into focus during the research process

(Study III). Interviewed paediatric cardiologists described the challenges and

expectations in dealing with the encountered complexity. For example, the diagnosis

process entailed breaking the news of the heart disease and dealing with the resulting

emotions of the children and parents (as well as the doctors’ own emotions), which

emphasized the dependence on the team doing its job. As part of the research interest

then shifted to the team (Study IV). The team’s challenge was to manage the

increasing complexity at both the family and organizational levels; this required

representatives of several professions and various team-development initiatives,

depending on the conditions of the team in question (Study IV).

Study groups

I. Ninety-seven children/adolescents with CHD, enrolled by means of consecutive

selection, were sorted into three categories with respect to complexity of CHD.

Group I included 42 patients with malformations requiring standardized

operations, group II included 20 patients with more complicated malformations,

and group III included 35 patients with very complex malformations. The patients

were compared with controls without heart disease, matched for age and gender

(Table 2, Fig. 3).

Table 2. The age distribution of the studied patients and of the age- and gender-matched

control group in Study I.

Age (y) n

0–4 42

5–9 29

10–14 10

>15 17


Children with CHD and their families Age and gender matched healthy

Children and their families

97

8

97

Figure 3. The patient and control populations in Studies I and II. In Study I, the patient

group consisted of 97 children and their families, of which four families participated in

Study II.

II. Interview data were collected from four families (Fig.3 eight people) on two

occasions, with a ten-year gap between the two interviews. The families were

strategically selected to fill the study criterion of representing all three levels of

complexity of CHD and several family members were selected (Fig. 4).

The interviewed families were as follows:

Family 1 mother and father were interviewed together in the first interview; the

mother alone was interviewed in the second interview

Family 2 both the mother and father were interviewed in the first interview; both

parents declined the follow-up interview, mainly because they were about

to be separated

Family 3 the mother was interviewed on her own on both interview occasions

Family 4 the mother and father together in the first interview and their son (sibling

to the child with CHD) in the first and second interviews


F

FB

M

F M

F M

1 1

1

2

2

11

Family 1 Family 2

Family 3Family 4

F = female, M = male, B = child

Figure 4. Illustrates the interviews in Study II. 1 = first interview, 2 = second interview.

III. We interviewed nine experienced paediatric cardiologists working at a tertiary

care centre for paediatric cardiology at a Swedish university hospital,

counselling families of children with CHD. One paediatric cardiologist also

participated in the focus group interviews (Fig. 5).


309

6

Figure 5. Paediatric cardiology teams/paediatric cardiologists.

In Study IV, 30 PCTs were involved in an inquiry, of which six teams participated in

focus interviews. Nine paediatric cardiologists were interviewed in Study III, one of

whom participated in the focus interviews.

IV. A national inventory/survey of all 34 PCTs was performed. Qualitative focus

group interviews were conducted in six PCTs selected by size and location

(Fig. 5).


RESULTS

Study I: Complexity

The complexity of the psychosocial situations of children and adolescents

with congenital heart disease

Aim

This study described the psychosocial situation of children/adolescents with heart

disease and their families.

Design and results

This study used questionnaire and quantitative data obtained from a group of 97

children and adolescents with CHD of different degrees of medical complexity and to

a control group.

In the patient group, 20% displayed one or more psychosocial symptoms evaluated as

severe, while in the control group the corresponding proportion was 1%. The three

grades of medical complexity differed regarding number and severity of psychosocial

symptoms: the children with the most complex CHD had the highest number of

moderate and severe symptoms, while those with the least complex CHD had mostly

mild psychosomatic symptoms. The most frequent symptoms in the whole patient

group in terms of the three spheres were healthcare and treatment-related needs in

the external sphere, family symptoms in the interpersonal sphere, and

mental/somatic/psychosomatic symptoms in the personal sphere.

Comparison of the patient and control groups indicated about the same overall

frequency of symptoms in the personal sphere, but that a larger proportion of the

patient group experienced psychological symptoms such as depression, anxiety,

loneliness, behavioural problems, and concentration problems, as well as

psychosomatic symptoms such as headaches and stomach pain, while more

children/adolescents in the control group had allergic, speech, and language

problems. Statistical analysis (Chi2 test, Fisher’s exact test) indicated that the

frequency of anxiety symptoms (p <0.002) and difficulties concentrating (p <0.001)

was significantly higher in the patient group than in the control group (unpublished

data). Headache problems were 15 times more common (p <0.0001) and stomach

pain twice as common (not significant) in the patient group than in the control group

(unpublished data). In the patient group, symptoms in the interpersonal sphere were

much more common (p <0.0001), especially when it came to family symptoms such

as crisis problems, parental desertion, and family interaction problems. Even in the

external sphere, a major difference was observed, with the patient group experiencing

more problems in most areas, including both clearly illness-related matters, such as

medical and rehabilitation needs, and matters relating to finances, insurance,

education, and transportation (p <0.0001).

R e s u l t s | 2 1

In conclusion, Study I elucidates the psychosocial complexity in children and

adolescents with CHD, indicating the need for structured psychosocial care

programme.

Study II: Coping

Facing bad news: a case study focusing on families having a child with

congenital heart disease

Aim

This study explored the experiences among parents and siblings to children with

CHD during the diagnostic phase and how family members dealt with the situation

over time.

Design and results

Interview data were collected from four families on two occasions, with a ten-year

gap between the two interviews. The analysis of the data revealed five main themes

and subsequent sub-themes

Table 3. Teams and subthemes revealed during the analysis.

Themes Sub-themes

Information about the diagnosis evoked overwhelming feelings

Reacting with stress, shock and confusion Feeling shame and guilt Feeling fear and anxiety Considering the situation as unfair Losing control

Ambivalence towards medical interventions and technology

Being dependent of others Lacking trust Feeling powerless and helpless Feeling lonely

Needing medical, social and emotional support and relief

Expecting medical and practical information Processing crises by counselling Asking for continuously support

Finding pathways to new perspectives Gradually getting additional information Well-functioning line of communication Professionals being present and giving time

Outlook of life and future change over time Gaining life experiences Getting increased self-knowledge and self-confidence Getting Increased acceptance Revaluing material conditions Having confidence in the future


Information about the diagnosis evoked overwhelming feelings: Our findings

indicated that receiving news of a child’s disease can be described as a turning point,

with effects persisting even after ten years. Regardless of the severity of the heart

disease diagnosis, the representatives of the studied families said that they were

initially shocked. Parents experienced feelings of guilt, anxiety, diffuse shame and

loneliness, and disappointment in life as such, perceiving the child’s disease as a

punishment. Surgical procedures and associated happenings were described as very

stressful. In addition to structural and relational circumstances, individual factors

proved very significant in shaping the studied families’ ability to cope, and these

factors also changed over time. What was common to our families was the stress this

news entailed, while their reactions and ways of evaluating and dealing with the

problems varied over time.

Ambivalence towards medical interventions and technology: The parents described

it as very stressful and mentally demanding to have to depend on others and not

always to understand what they were experiencing. They said that it was sometimes

difficult to have trust in the consequences of the major therapeutic interventions and

efforts required.

Needing medical, social, and emotional support and relief, finding pathways to new

perspectives, and outlook on life and future change over time: When the initial

shock at the diagnosis had abated and the parents had begun to formulate questions,

they experienced a growing need for contact with health care professions and

information. Access to social support from people outside the healthcare system was

highly variable. In both the short and long terms, well-functioning communication

between the families and medical personnel was essential. PCTs’ willingness to

communicate and to structure information in useful forms gained the families’ trust,

and the doctor’s professionalism in breaking the news was crucial. For all the

children with heart disease, the interviews revealed that the purely medical course of

events over the past decade was more positive than the parents had initially feared.

From a longitudinal perspective, we found effects even after ten years. The

interviewees said that their feelings had changed over time and that they now felt less

anxiety. Despite marked stress, living with and caring for a child with heart disease

proved manageable for the studied families.

Study III: Profession

Breaking bad news: an interview study of paediatric cardiologists

Aim

This study examined the approach of paediatric cardiologist in informing and

communicating with the family of the CHD patient.

R e s u l t s | 2 3

Design and results

Interviews with paediatric cardiologists focused on their encounters with families of

children with CHD. The interviews dealt with parent and patient counselling

throughout the cardiologists’ careers, including the counselling of parents whose

infants were diagnosed with complex CHD requiring some kind of interventional

procedure.

The interviewed doctors unanimously stressed the importance of good

communication when meeting with families, but their strategies for facilitating such

communication varied. Doctors’ strategies for meeting with families varied in terms

of structure, relationship, and degree of formality. Meetings ranged from a very

structured information meeting to a more flexible format, from relying on formal

guidelines to being shaped by the doctor’s personal experience, and from merely

providing factual information to accepting the emotionally charged nature of such

meetings (Table 4).

Table 4. Overview of the results from the study of paediatric cardiologists.

Method of providing information Consensus about importance of information From solid structure – flexible form Absence of training in methodology Guidelines are not applicable

Emotional positions Broad range from close and empathetic to more distant and detached A “middle way” was most common

Building confidence Seen as essential Strategy for doing so varied Building trust was part of the process Confidence and trust had to be mutual

Interaction Draw and talk Repetition Consider recipient ability and skill Continuity is important Both individual and family/group perspectives needed

Success and failure Existence of trust is crucial to success Communication breakdown causes failure

Strategies for handling the situation

Developing dialogue Mentoring Working on common values Finding a common strategy Providing guidance Allowing space for reflection Team development


Method of providing information: There was broad consensus among the

interviewed cardiologists that it was essential to provide complete and accurate

medical information. When it came to information provision, the doctors’ strategies

ranged widely from formally structured to more flexible. The cardiologists agreed

that they lacked training in information provision/conversational methodologies.

There was near consensus that general guidelines were not always applicable.

Emotional positions: Overall, the emotional positions employed ranged widely from

empathetic and close to more distant and detached; most doctors’ chose a “middle

way without being too close”, while some vacillated between closeness and distance.

Building confidence: Creating confidence in the patients and their families was

essential to the doctors’ information provision strategies. There was widespread

agreement that building trust was essential, and the cardiologists used various

strategies for doing so.

Interaction: Most of the doctors drew sketches, and many used repetition in meeting

the information needs of their patients and parents in a stressful situation. Most

considered the recipients’ communication ability when interacting with them.

Continuity between the physician and parents was considered important; likewise, it

was important to consider both individual and group perspectives.

Success and failure: Failure was described as resulting from communication

breakdown, while success arose when doctors and patients/families created mutual

trust.

Strategies for handling the situation: Existence of a common strategy, provision of

guidance, allowing space for reflection, and team development were mentioned as

tools for managing the complexity surrounding the child with CHD.

In conclusion, this study found that paediatric cardiologists stress the importance of

conveying bad news in suitable ways, building mutual trust, and adopting various

emotional positions in relation to the families. All cardiologists wished to improve

their ability to handle such situations and expressed a need to work on common

values, reflection, and education.

Study IV: Teams/PCTs

Teamwork in Swedish paediatric cardiology: a national exploratory study

examining function and dynamics

Aim

This study described the organization and function of PCTs in Sweden, and sought to

deepen our understanding of working in a PCT

R e s u l t s | 2 5

Design and results

A national survey of all 30 PCTs was performed by means of a questionnaire.

Qualitative focus group interviews were conducted with a total of 29 members of six

PCTs selected by size and location. One cardiologist participated in the focus

interviews (Fig. 5).

The questionnaire was answered by nurses in 17 of 26 PCTs, and the nurse was the

coordinator in 17 PCTs. All teams emphasized the need for consistency in dealing

with the child with CHD and the importance of analysing the team’s joint

responsibility for medical and psychosocial care in order to decide on a shared

holistic approach. All PCTs estimated, using a seven-point scale (1 = low estimation,

7 = very high estimation), the degree of teamwork experienced: one team rated the

teamwork at 5 points, eight teams at 6 points, and 17 teams at 7 points.

The complexity of the cases at both the family and system levels obviously required a

team of representatives of various professions. The PCTs tried to work in a structured

way, but team structure and composition differed, and problems arose because of the

working conditions. The cardiologist’s work, time, and interest were essential to case

progress. The complexity and constant change of the children’s cardiac care led to a

need for more cooperation within and between teams, and among the teams, team

physicians, child and adolescent psychiatry (CAP) services, habilitation organizations

(HOs), and school systems. All teams experienced more or less serious problems

communicating constructively with CAP and the HOs.

In conclusion, managing the continuously changing needs of children with CHD and

their families called for organizational alteration and action plans in PCTs. The teams

need to further develop their leadership capacity, resource coordination,

collaboration with organizational support, continuing education, and access to

coaching.


DISCUSSION

Children and young people with cardiac disease, together with their families,

represent a growing and somewhat new diagnostic group for which we cannot draw

generalized conclusions based on experience with other diagnostic groups. The

specifics of this chronic disease group, such as the complexity of their situation,

experience, and the invisibility of their handicap, with various crucial symbolic

implications, has consequences for their psychosocial care and treatment (17).

Emotional consequences, communication, information, and support are essential for

these children, parents, and families as well as for the professionals and PCTs

treating them. In an era of intensifying discussion of priorities and economic

austerity, it is even more important to analyse the consequences of an insufficiently

holistic approach. Provision of necessary resources for the psychosocial needs of

these children and adolescents and their families must be continuously monitored

and developed (2).

Complexity

A growing body of literature addresses questions concerning the QoL and living

conditions of children with CHD (17, 96–98). Latal et al. (17) conducted the first

systematic review of the last 18 years of literature on the psychological adjustment

and QoL of children and adolescents with CHD following cardiopulmonary bypass

surgery, noting methodological variations and restrictions that make it difficult to

compare studies and draw general conclusions. In this respect, there is a complexity

in the literature that must be addressed. As in this thesis, Latal et al. (17) note that

QoL is frequently used as a concept and methodology in understanding and assessing

the psychosocial situation in CHD (99); it represents a multidimensional construct

that includes social dimensions, stress, and emotional and cognitive functions. The

findings of our and other studies from the QoL and HRQoL perspectives can be used

to deepen our understanding of how to develop interventions for children with CHD

and for their families, with the aim of reducing psychosocial morbidity (100).

Neuner et al. (101) demonstrated in a 2011 study that sense of coherence (SOC) was

associated with overall wellbeing and with QoL sub-dimensions for adults with CHD.

Similarly, Moons et al. reflected on the correlations between a well-structured or

“high” SOC and the opportunity to improve QoL (102).

We developed a model illustrating the overall psychosocial situation facing children

with CHD and their families; this model has been useful in analysing and highlighting

various aspects of the psychosocial complexity of this patient population. In Study I

of this thesis research, the complexity of the psychosocial situation of child CHD

patients and their families was illustrated by the high frequency of symptoms in all

areas, i.e., the personal, interpersonal, and external spheres.

D i s c u s s i o n | 2 7

Personal sphere

In the personal sphere, children with CHD had significantly more problems with

depression, anxiety, and concentration as well as more behavioural problems.

Increased internalizing and AD/HD problems are seen in recent studies of children

with CHD (17). In our study, patients with CHD had a statistically significant higher

frequency of mental health and psychosomatic symptoms than did age- and gender-

matched controls.

The study group includes children who have been in a heart-lung machine, which

seems to be a risk factor for AD/HD symptoms (103). The stress of invasive surgical

procedures, hospital care, and the impact of having heart dysfunction are important

both symbolically and in reality and are thus important determinants of mental

health and QoL outcomes (98, 104).

A growing literature that highlights questions concerning the increased frequency of

mental health symptoms in children with CHD, as related to various risk factors,

supports the results of Study I (104, 105).

Interpersonal sphere

The most severe problems experienced by the studied CHD patients and their

families were seen in the interpersonal area, highlighting the family burden of living

with a child with CHD (106). This was even more obvious in comparison with

controls, who experienced significantly fewer family-related problems.

These results indicate, as Sarajuri and Brosig demonstrate (107, 98), that severe

congenital heart defects increase parenting stress. The described family symptoms

can also influence parent–child interaction, making parents less able to care for

children with CHD, rendering the child more vulnerable (16, 108-111). The findings

raised questions about whether the children’s, adolescents’, and families’ QoL has

decreased, but not in all sub-domains (17, 98). Like this thesis, Latal and Brosig’s (17,

98) demonstrate that these children may have behavioural problems. This finding

can mainly be associated with parental descriptions of the situation of the children

and family. Goldbeck and Melches (15, 16) and Solberg (112) find that parental QoL

was less associated with the severity of the child’s illness than with social risk factors,

such as being a single parent.

External sphere

Healthcare and treatment needs were frequent external sphere needs, as were, for

example, social service, financial, and travel/transportation assistance. Connor et al.

reached similar conclusions, examining the level of complexity in terms of the need

for resources and interventions from the healthcare system, the environment, and

society (31). Other authors exemplified this in a study from neonatal care (113). Care

based on a professional approach at all levels has long-term positive consequences for


children/young people with CHD and their families and helps eliminate the risk of

psychosocial sequelae (114, 115).

We identify a complexity of which is supported by the literature (26, 27, 31, 113, 114).

One approach to managing and structuring complexity is offered by the International

Classification of Functioning, Disability and Health (ICF), published in 2001, a

paediatric version of which has been available since 2007 (ICF-CY). The ICF-CY is

based on the diagnosis, but takes account of the “big picture” relating to daily

functions, symptoms, activities, and participation, as well as to household conditions

and social factors (33). The model we developed can be viewed as an earlier attempt

to showcase the psychological complexity of a specific patient population.

Coping – Stress

The difficult situation that the studied parents found themselves in when they

learned the news of their children’s illness changed their lives. We describe this

situation as a turning point (116) in life, but could also have chosen to describe it

using Strauss and Glaser’s (117) concept of “status passage”. It is hardly surprising

that a negative and decisive turning point in life, such as learning that a child suffers

from a serious heart disease, generates problems for the entire family, but our data

also indicate some positive trends, especially from a longitudinal perspective.

As demonstrated in Study II and other studies, being informed of their children’s

congenital heart defects commonly engendered stress and strain for the patients’

families, both at the time of being informed and over time (26–28, 111). Like various

authors (26, 27, 118), we find emotional reactions such as anxiety, fear, and a

changed way of valuing and dealing with problems, as well as a sensed burden of

responsibility and financial uncertainty (31). The present findings can be compared

with those of Menahem and Green (111, 119), who state that healthcare personnel, for

example, PCTs, have to be aware of and identify psychological ill health as a risk

factor for both parents and children. Parental descriptions of how the initial post-

diagnosis period with the child was fraught with anxiety and entailed many life

adjustments coincide well with the results of an interview study of parents of children

with cancer (120).

Arafa (121) found that 98% of parents of children with CHD worried about their

children’s health and future. In our study, especially in the early period of illness, the

parents describe worrying about the future; after 10 years, they still experienced

occasional bouts of worry and concern.

In addition to structural and communicative relationships relevant to the possibility

of dealing with the situation, individual factors relating to the parents and family also

played a role (122). These factors also appeared to change over time. Parental

wellbeing also appears to be related to the psychological adjustment of their children

(17, 123).


The parents describe finding a pathway when having a child with CHD and

summarize their previous life in terms of major life experiences that can strengthen

the couple’s relationship (122).

As an increasing number of children with CHD survive and require life-long follow-

up (14), it also becomes important to understand the mechanisms underlying the

behaviour of teenagers with CHD and their families (123). Zahmacioglu et al. (124)

believe that specific knowledge of coping strategies is needed for this patient

population and their families. Lazarus’ coping theory (35) underscores the interaction

between problem-oriented and emotion-focused strategies for action, and indicates,

as do our results, that individual factors are often crucial determinants of people’s

ability to orient themselves in situations of extreme stress. The present results are

consistent with Lazarus’ transactional stress model (50), in which emotions play a

dominant role, especially the interplay of emotions with cognitive assessments of the

action context and of the action strategies that therefore become relevant. We have

focused on three aspects of what Lazarus terms secondary appraisal. The first aspect

concerns the evaluation/assessment of who is responsible for the problem and to

what extent, and includes feelings of guilt and shame, as well as positive values such

as pride. Of particular interest here is the parents’ assessment of their own efforts.

The second aspect involves assessing the opportunities for dealing with the problems,

i.e., coping potential. Even here, we focus on the parents’ own opportunities as well

as those of the professionals, mainly doctors, with whom they come into contact. The

third aspect involves future expectations, an approach found to play a major role in

the communication patterns that we described. Secondary appraisal represents an

ongoing cognitive and emotional assessment of the situation in which the families of

children with CHD found themselves during the decade we monitored. This finding is

consistent with those of Lawoko (125), who describes how parents of children with

congenital heart defects initially experience crisis in the first year after being

informed of the diagnosis. Like our subjects, Lawoko’s subjects describe emotions

such as anxiety and anger, but these emotions change and improve. Lawoko’s results

also indicate that parents develop coping strategies and resilience. The families

examined in the present study display “resilience” that may depend on professional

support and networking, as Tak and McCubbin (126) also demonstrated. One

example of the resilience (127) of a family living with a child with CHD is presented

by Wray and Sensky (122), who describe how parents felt that their relationship had

strengthened during the period when their children underwent cardiac surgery.

Before surgery, these parents had stable relationships, which became even stronger

afterwards. Our study demonstrated that, despite marked stress, caring for a child

with CHD proved manageable for the studied families. The studied parents also

described how the shared experience of dealing with their child’s illness positively

developed their relationship.

In line with the results of other studies (17, 19, 128, 129), our results also indicate that

psychosocial complexity creates a need for psychosocial support for children and

adolescents with CHD and their families. Several studies illustrate the impact of this


complexity on daily life, examine mechanisms for coping with the situation, and

emphasize that support is needed and requested (104, 105, 126). Parents described

needing to fight for their children’s and their own rights. They described the

challenges of obtaining information in stressful situations, and said that they felt

responsible for informing others about what it means to live with heart disease in the

family. Parents describe receiving support from grandparents and social networks

(114, 115) and rating the support of healthcare professionals, the PCT, and the

paediatric cardiac clinic as very significant (114, 125, 128). Well-functioning

communication with the healthcare system (in this case, the PCT) in both the short

and long terms provided parents with essential help in managing the situation. As it

is paediatric cardiologists who often break the news of a child’s CHD, the

professionalism of their approach seems to be crucial to the families (130-135).

Profession

A key moment is when the doctor informs parents of their child’s heart problem

(2007). Like Menahem (136), we find that parents view this meeting, which sets the

course of their ongoing interaction with the healthcare system, as crucial and often

prolonged. Against this background, we found it important to study the attitude of

the paediatric cardiologists. The physicians came to play a central role in our study,

but their position in relation to the family could be assumed to be generalizable to

describe the professional aspects of the encounter with children with CHD and their

families.

While the interviewed doctors agreed on the importance of this initial encounter,

their views varied as to the requirements for a good encounter. One important

characteristic of the encounter is the degree of empathy displayed by the doctor, and

the question raised here is whether empathy is consistent with the professional role

of doctor (137). Is sharing how other people (in this case, the parents) experience

events a relevant response to the expectations these people have of the doctor, or

does it fall to other professional roles, such as the social worker or nurse?

Nevertheless, study III focuses on the role of the doctor and how it manifests itself in

the patient encounter. In the preface to their book Profesjonsstudier [Professional

studies], Molander and Terum (138) divide research into professions into three

problem areas. The first relates to qualifications, i.e., how to learn the profession.

While the cognitive conditions of the medical profession are well entrenched in the

education system, its emotional aspects receive much less attention. A second

problem area concerns the status of knowledge, and here we mainly underscore the

needs of theoretical versus clinical knowledge. The third area concerns the practice of

medicine and how to deal with the often conflicting demands and expectations aimed

at the profession by various parties, especially by patients (138).

One difference in attitude regarding the various approaches to the encounter with the

patient/family is the perception of one’s own effort. Conventional theory about

professionals assumes that professionals block their emotions, or at least, in the


present case, do not allow them to affect encounters with children and families (139).

Here we found significant variation. While some professionals saw themselves as

dominant in the encounter, others viewed the parents as clients and themselves as

providers of what was expected. A difference was even apparent between the

attitudes that the meeting should be characterized either by a firm and stable

structure or by a flexible approach in which the doctor tries to read the parents’

reactions and then adapt to them. However, information and communication

comprised a key concept for all interviewed doctors, and there was a clear

understanding that “communication breakdown” represented failure in their work.

One clear result was that doctors felt that the most important factor contributing to

the sense of a job well done was the creation of trust (140).

By definition, being a doctor means being expected to be professional. The medical

degree and license promises this professionalism and in most cases patients expect a

professional encounter in connection with consultations and treatment (138). What

really provides this legitimacy is the belief that it is based largely on science and

proven experience. Professional practitioners and their experience can be considered

in terms of Carr-Saunders and Wilsson’s (141) classic position on the issue of the dual

role of the professional as mediator and interpreter. In the patient encounter, the

doctor is expected to provide accurate information about diagnoses and risks, and to

translate the scientific language into lay terms so that patients understand what is

involved.

People’s expectations of professionals reflect the idea that a modern society entails

the differentiation and extensive division of duties (59). Being professional requires

objectivity, knowledge, and the attitude that all clients should be treated equally.

Moreover, the status of the professional must be acquired, mainly through education.

All this is expected to make the patient confident that he or she will receive a proper

diagnosis and treatment. Malhotra et al. (142) discuss the ethical issues underlying

the doctor’s professional counselling task, although they focus particularly on ethical

dilemmas in foetal management from a cardiac perspective. Malhotra et al. cite actual

cases to highlight the increasing counselling challenges facing physicians. Here it is a

question of reconciling the expectations that professionals face with their own

personalities (59).

The studied doctors consistently said that medical school spent little time on this

issue and several respondents said they were poorly prepared for their first patient

encounter, which caused them to be nervous and uncertain. All interviewees stressed

the lack of appropriate education in this regard and requested more training rather

than simply guidelines. Rosenbaum (143), in a review study, and Fouron (134) and

Fallowfield (135) discuss aspects of this issue and reiterate the necessity of being

aware of the importance of developing this learning area. Another striking finding

was that many cardiologists emphasized the importance of reflection. In line with

this, many authors have stressed that the practitioner should reflect on current

professional perspectives (136, 144, 145).


It was striking that the cardiologists, besides individual approaches to improving

communication, also emphasized the importance of team meetings and of developing

teamwork, particularly in PCTs.

Teams/PCTs

Earlier studies have demonstrated that both families and cardiologists often value the

local PCT as a key central unit (146), stressing that teamwork needs to be an

important part of the continuum of care to meet the complex needs of children with

chronic disease and their families (147). In study IV, we arrived at the same findings,

i.e., that the knowledge needed to best treat a child with CHD and deal with his or her

family is now so extensive that appropriate treatment requires a team comprising

representatives of various professions. We also found that, although the goal is to

work as a team, there are ambiguities in terms of who is responsible for what aspects

of care and for relationships with other societal actors. There is a need to clarify and

manage the mission of caring for the child with CHD in relation to the increased

complexity and constant change in healthcare and society. Conceptual robustness is

required if a team is to respond to this need at the professional level, with respect to

both the child and his or her family (148, 149). If the professions want to collaborate

in teams to provide the desired holistic approach, the organizational answer is

interprofessional teams (150).

In our study, we identified a great desire to work in teams and a willingness to take

on structural challenges, but the teams nevertheless encountered difficulties and the

discrepancies with ideal ways of working created frustration. For teams to work

efficiently and cope with both current and future missions requires deliberate efforts

to build robust and collaborative cultures, which include further development of

cooperation in workgroups and of workgroup management (151, 152). Factors of

importance for PCT ability to work, as identified in Study IV, were coordination of

team resources, support, continuing education, and access to professional

management consultants.

Dependence on the physician and the necessity of a physician being present in the

PCT were expressed by all interviewees, and it was evident that the physicians’ lack of

availability limited the PCTs’ ability to work smoothly. Thylefors et al. (152, 73)

discuss how lack of time weakens the physician’s ability to participate in the team;

this time pressure means de-emphasizing care conferences, which reduces the

meaning of the interdisciplinary team.

The studied PCTs were formed especially to treat the children with CHD and their

families, and the teams and their internal relationships were built through social

contact within the teams. The teams provided an unexpected description of their

internal communication, presenting an image of integrative cooperation and few if

any internal conflicts. This presentation could be explained in two ways: either the

teams constituted genuinely mature groups, or they sought to project an idealized

group self-image (152). Thylefors also state that conflicts are essential in


interdisciplinary teams; however, the studied PCTs could constitute properly

functioning working groups, i.e., “trans-professional teams” (75, 76,) in which

conflicts are rare. Earlier studies have demonstrated that lack of organizational

structure, difficulties prioritizing, and poor meeting structure could impede

teamwork (153). Structural and organizational limitations must be exposed at all

levels and eliminated, so that the PCTs’ ability to work in a process and meet

continuously changing needs can develop. Cashman (154) describes this in a case

study of integrated teams in primary care, which stated that organizational structures

and incentive programmes must be related if efficient team function is to be

sustainable.

The studied teams mentioned no explicit leaders. In some teams, the person who

initiated the team meetings served as the temporary leader, while in others, the nurse

and the physician implicitly assumed leadership roles. Like McCallin (155), we want

to raise the question of more directive leadership, and its consequences for PCT

structure and development.

The rapid expansion of this field has led to proliferating knowledge on the part of the

various professions involved in the rehabilitation process, in turn increasing the need

for efficient interaction between experts in different fields (19, 156). The PCTs

evidently had some problems communicating constructively with CAP and the HOs,

which sometimes led to conflicts between individuals, professions, and units.

Cooperation between these entities must be managed and further developed (157).

Dialogue is an important and characteristic cooperation tool in interprofessional

teams (153). Conflict management and constructive communication give a broad

perspective on team functioning, and our results indicate a need to improve team

communication by allowing time for dialogue, problem solving, and knowledge

sharing. As the needs of the children with CHD and their families are increasingly

complex, clinical experience indicates that collective reflection is necessary in PCTs –

there is a need for a “forum for reflection”. Dialogue and discussion concerning

objectives, common understanding, ethics, and collegial support are crucial (157).

There is a need for joint reflection in the local PCT, and in regional and international

paediatric cardiology centres as well. The establishment of an ongoing network of

PCTs may result in national collaboration for research and clinical improvements for

children with CHD.


METHODOLOGICAL CONSIDERATIONS AND LIMITATIONS

Quantitative method

At time of planning this study, we were not aware of any existing instrument

capturing the complexity of psychosocial factors in children/adolescents with heart

disease and their families. Lindström replied positively to the suggestion to put his

theory into practice (34). We found it obvious that more specific disease-adjusted

methods for elucidating psychosocial complexity were needed (158, 159) Drotar et al.

(160) explain how QoL assessments can be used for many purposes, and these

authors describe both the necessity and complexity of understanding and describing

the situation experienced by chronically ill children. In Drotar et al., we found

support for the first study describing the psychosocial situation of these patients,

using a new psychosocial inventory. Guided by previously validated instruments

(161), our aim was to characterize the complexity related to children with cardiac

disease using an assessment instrument to assess/describe their psychosocial

situation (162–164).

As Goldbeck and Melches (15) state in the introduction to their 2005 study, we also

found, at the time of the first study, that many instruments developed for measuring

QoL in children lack the perspective of the child with CHD within his/her family.

In 2001, the International Classification of Functioning, Disability and Health (ICF),

was developed. A paediatric version of this, ICF-CY, has been available since 2007

(33). The ICF-CY is based on the diagnosis, but views the “big picture” relating to

daily functions, symptoms, activities, and participation, as well as to household

conditions and social factors (33). HRQoL scales adapted for children with cardiac

disease are now also available, which was not the case at the time that our study was

planned (39, 40).

One general limitation is that the instruments used in study I and study IV were

developed especially for these studies and have not been psychometrically validated.

The untested reliability of the inventory method could be seen as a weakness, but, in

the method’s defence, most parts of the instrument derive from pre-existing,

validated instruments (16, 164). Comparison with a matched control group and the

relatively extensive material increase the reliability of the instrument’s results. That

the instrument was clearly useful to the well-informed research group that used it,

further attests to its utility. Minor limitations of the method are that it requires an

experienced clinical social worker for its application and that sociodemographic

covariates such as socioeconomic status, education, and profession are not included

in the inventory.

M e t h o d o l o g i c a l c o n s i d e r a t i o n s & l i m i t a t i o n s | 3 5

Qualitative method

The focus of study II was on exploring the experiences of parents and siblings of

children with CHD during the diagnostic phase and on following how family

members dealt with the situation over time. The use of a broad opening question in

the interviews allowed the parents and siblings to choose how to describe their “way

of living” close to a child with CHD. The interviewees were asked to reflect on their

experience of this situation. The judgment of trustworthiness findings should be

based on credibility, dependability, and transferability (165, 166). One aspect of

credibility practiced throughout the thesis work was open dialogue between the

researchers: the authors of the studies discussed the data interpretation to ensure

that judgments about differences and similarities were consistent (94).

We believe that the samples achieved an acceptable level of redundancy, and that the

participants in the constituent studies of this thesis have been sufficiently covered in

the material. Though it could be argued that the numbers of participants in the

studies were small, trustworthiness in a qualitative study is gained more by the

richness of each interview than by the sample size per se (167–169). That being said,

the sample size in Study II did make it difficult to identify differences between

families attributable to the level of disease severity, sociodemographic factors, or

differences in access to resources relevant to their coping behavior.

In Study III, we interviewed a small number of paediatric cardiologists working at the

same Swedish department of paediatric cardiology. Though the results are local and

limited, with the theoretical framework serving as background, we find the

conclusions generally applicable. Another limitation is the lack of gender balance

because, at the time of the interviews, all consultants in the chosen department were

male.

The results of Study IV may also be of limited applicability because Sweden is a small

country; on the other hand, the survey conducted is a national one including a large

proportion of existing Swedish PCTs. As our aim was descriptive rather than

theoretical, we believe the sample achieved an acceptable level of reliability.

Content analysis can be carried out with different levels of abstraction. One approach

is to view the manifest content, i.e., what is overtly expressed in the text, which is the

most common approach in quantitative research. However, we chose a different

option for the constituent studies of this thesis, also analysing the latent content,

which entailed interpretation. Graneheim and Lundman (92) believe that some form

of interpretation is always involved, though its depth may vary, and other authors

describe other similar approaches. It might have been possible to apply an approach

similar to that of Downe-Wamboldt (95), who recommends formulating preliminary

categories after a first, superficial review of the material and then encoding all the

text.

Clearly, there are different ways of thinking and it became ever more apparent over

the course of the research process that it was impossible to go through the process


directly from beginning to end, but that progress was instead iterative between the

various stages (92).

One measure of the reliability of an analysis is to openly describe the analytical

method and how it was implemented, how the data were collected, and the context in

which the data were collected (95). Regarding the follow-up portion of study II, the

material was limited in that interview material was available from only two occasions

involving the same people, which obviously reduces the amount of information

produced by the study.

We established internal validity – i.e., the truthfulness in representing the reality of

the participants – by carrying out a “member check” as suggested by Lincoln and

Guba (165). Member checks do help improve the accuracy, credibility, validity, and

transferability (also known as applicability, external validity, or fittingness) of

research results. In many member checks, the interpretation and resulting report (or

a portion of it) are given to members of the sample (informants) who check the

accuracy of the work. In studies III and IV, we used the member check to obtain

informant feedback or respondent validation. Their comments serve to check the

viability of the interpretation and were described in the studies.

Early in the research process, we found that a combined quantitative and qualitative

approach best suited the overall purpose of the study. Quantitative and qualitative

methods do not need to stand in opposition to each other; instead, we viewed both

approaches as equally valid tools for better understanding the context we wanted to

explore. Combining methods gave a broader database and, consequently, a more

secure basis for interpretation. We saw that combining various methods would

provide benefits such as increased validity and greater confidence in the analysis,

facilitating the development of a nuanced and holistic picture of children with heart

disease and of their general situation (165, 170).

G e n e r a l s u m m a r y | 3 7

GENERAL SUMMARY

In conclusion, the inventory method can differentiate the three grades of medical

complexity, in terms of number and severity of psychosocial symptoms. The

inventory of the personal, interpersonal, and external spheres elucidates the

psychosocial complexity of symptoms and creates a basis for understanding the need

for psychosocial support in children and adolescents with CHD.

Receiving news of a child’s heart disease can be described as a turning point, a central

event in the lives of the affected families. What is common to the studied families is

the stress this news entails, while their reactions and their ways of evaluating and

dealing with the problems vary over time. Despite marked stress, living with and

caring for a child with heart disease proved manageable for the studied families. In

both in the short and long terms, well-functioning communication between the

families and medical personnel was crucial. Staff readiness to communicate by way of

their availability, continuity, manner, structuring of information, and supportive

behaviour created a feeling of security, leading to trust on the part of the families of

the children with heart disease.

Among paediatric cardiologists, we found unanimous emphasis on the importance of

properly communicating bad news: they agreed on the significance of trust and

confidence in relation to the families and on the use of various emotional positions

when breaking the news of the diagnosis. A common aim was to achieve better skills

at handling the situation, by means of education, coordinating efforts through joint

reflection, and sharing experience. To meet these needs, there is a need to develop

teamwork and for medical education programmes to develop the skills of medical

students and residents at delivering bad news.

Swedish PCTs aim and desire to work in a structured way, usually with a nurse or

contact nurse as coordinator, though team structure and composition differs between

units. The challenge is to meet and manage the increasing complexity of paediatric

cardiac cases at both the family and system levels, which requires a team of

representatives of various professions. These teams need leadership, resource

coordination, support, continuing education, and access to coaching. The physicians’

lack of availability limits the PCT’s ability to work smoothly and to meet the

continuously changing needs of children with CHD and their families. Managing this

constant change calls for organizational change and action plans. There is a need for

fora to stimulate dialogue and joint reflection in local PCTs and at regional and

international paediatric cardiology centres as well.


CHALLENGES FOR THE FUTURE

- To meet and manage the increasing psychosocial complexity for children with

CHD and their families.

- To further develop psychosocial care programmes including methods for

describing and assessing the patients’ and their families’ psychosocial needs from

a bio-psychosocial perspective– taking into account the chronicity of CHD.

- To actively participate in the process of developing and apply patient-reported

outcome measures (PROMs).

- Fora for individual and group reflection/supervision/coaching should be offered

to doctors, nurses, and paramedical staff involved in treating and caring for

children with CHD.

- To create conditions for external and national cooperation/collaboration, e.g. by

communicating and taking advantage of a recently introduced national video

system.

- To raise the awareness of the psychosocial complexity of children with CHD, to

stakeholders such as politicians, administrators at various levels, lobbyists,

community and healthcare leaders and the public.

A c k n o w l e d g e m e n t s | 3 9

ACKNOWLEDGEMENTS

First of all, I would like to thank

All the participating children and their families/parents who taught me

something about life and living.

The professions that were available, shared thoughts and professional experiences.

A lot of people have contributed to this work. In particular I would like to thank:

Annika Rydberg My main supervisor for patiently, but firmly, introducing me to

the field of research. With your width of knowledge, common sense and enthusiasm,

you are a role model.

Bruno Hägglöf My co supervisor, for opening a world of research and for valuable

practical support.

Olle Hernell and Magnus Domellöf as previous and present Chairman of the

Division of Pediatrics.

Lars Dahlgren for helping me deepen my understanding of qualitative method and

for valuable discussions over a cup of coffee and a tasty biscuit.

Ulla H Graneheim for sharing your competence in qualitative methods.

Birgitta Bäcklund for turning the word of “kappa” into the substance of a book.

Ina Höjmark, Mona Strid, Barbro Nilsson, and Dag Teien at the Pediatric

Cardiology Clinic in Umeå thank you for lots of laughter and many years of

teamwork.

Kirsi Reijonen my colleague and friend. For long lasting friendship, for listening,

for a dialogue that brought me forward and for cooperation in the research fields “the

art of questioning” and management/leadership agility.

Eva Tingåker Johansson for friendship, a warm heart and encouragement along

the way and for bringing me outside of the academic world reminding me of rest of

life.

Annica Anca Elmberg, for your creativity, for your illustrations filled with

meanings and thank you for patience when I have to “cut” them.

Anneli Kero, Elisabeth Rhodin, Katarina Bergström, Ing-Marie Marklund

colleagues thank you for cooperation and friendship during all years.

Ulla Svensson for providing me a “home away from home”.

Ola, Jacob, Lydia and Pontus My family: The essence of my existence, for love,

support and patience.


This thesis has been supported by grants from: Swedish Heart Child Association

(SHCA), Samaritan Foundation and fundings from the Northern Country

Councils Cooperation Committee.

For funding throughout the years leading up to completion of this thesis I would like

to acknowledge Division of Pediatrics and Division of Child and Adolescent

Psychiatry, Department of Clinical Sciences.

R e f e r e n c e s | 4 1

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