New insights into family functioning andquality of life after pediatric livertransplantation

Liver transplantation is now standard treatmentfor children with ESLD. Medical improvementsfollowing pediatric liver transplantation are wellestablished, and long-term survival is the rulerather than exception with patient survival ratesat five-yr post-transplant as high as 82% (1, 2).With up to 15% of all liver transplants per-formed on pediatric patients, long-term implica-tions are receiving increasing attention (3). Earlyfocus on recipients� health, physical growth, andcognitive development preceded interest in QOLoutcomes, with much research producing prom-inent findings of QOL differences between pedi-atric transplant recipients and non-transplantchildren, and QOL improvements from pre- topost-transplantation (4, 5).

The QOL of pediatric liver transplant patientshas received widespread attention, yet the well-being of their families is less often investigated,leading Alonso et al. (2003) to call for betterassessment of the effect of transplantation onfamily functioning. In addition to the emotionalconsequences of a diagnosis of ESLD and under-going the transplant process, parents mustquickly begin to accommodate an ill child byreshaping daily family routines and reorganizingresources, activities, goals, and priorities (6). Inthis study, family functioning refers to thefrequency of disruption to usual family routines,effectiveness of family communication and prob-lem solving, family cohesiveness, and how wellfamily members get along (7). This focus reflectsthe importance of consistent family routines asintegral to a child�s well-being. Regular familyroutines teach children about individual andfamilial patterns of behavior while establishingimportant developmental and social pathways.This offers opportunities of positive psychological

Denny B, Beyerle K, Kienhuis M, Cora A, Gavidia-Payne S, HardikarW. New insights into family functioning and quality of life after pedi-atric liver transplantation.

Abstract: Thorough research of the medical aspects of pediatric livertransplantation has given way to recent interest in the impact of thetransplantation process on the QOL of recipients and their families. Inthis cross-sectional study, we compared the family functioning andQOL of children (n = 30) aged between three and 16 yr (M = 10.10,s.d. = 3.62) who had received a liver transplant in the previous 1–12 yr(M = 5.31, s.d. = 3.44) with non-transplant children (n = 33), asreported via parent proxy. Results showed that parents of pediatric livertransplant recipients made significantly more adjustments to familyroutines to accommodate their children, particularly in relation tochildcare. Impaired family functioning was also found to be associatedwith decreased QOL. These preliminary findings of relative deficits infamily functioning may inform psychosocial interventions to assistpediatric liver transplant patients and their families. Further investiga-tion beyond a single-center study incorporating subjective informationfrom pediatric patients and their parents is recommended.

Bianca Denny1, Kathe Beyerle2,Mandy Kienhuis1, Ancuta Cora1,Susana Gavidia-Payne1 and WinitaHardikar2

1Discipline of Psychology, RMIT University,Melbourne, Victoria, 2Department ofGastroenterology, Royal Children�s Hospital,Melbourne, Victoria, Australia

Key words: pediatric liver transplantation – familyfunctioning – family adjustments – quality of life

Bianca Denny, Discipline of Psychology, RMITUniversity, PO Box 71, Bundoora, Victoria, 3083,AustraliaTel.: +61 3 9925 7376Fax: +61 3 9467 7503E-mail: bianca.denny@rmit.edu.au

Accepted for publication 7 May 2012

Abbreviations: ESLD, end-stage liver disease; FAQ, FamilyAccommodations Questionnaire; manova, multivariateanalysis of variance; OTC, ornithine trasncarbamylasedeficiency; QOL, quality of life; RCH, Royal Children�sHospital.

Pediatr Transplantation 2012 � 2012 John Wiley & Sons A/S.

Pediatric TransplantationDOI: 10.1111/j.1399-3046.2012.01738.x

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experiences that contribute to self-esteem, suchas pleasure, attachment, and competence (6).Further, frequent change and unpredictable dailyroutines have been identified as unhelpful tochildren, as the sustainability of family life iscompromised, affecting the well-being of allfamily members (8).Limited investigations of families following

pediatric liver transplantation have focused onseveral aspects of family functioning. No differ-ence in family dysfunction was detected betweentransplant families and a community-based sam-ple (9), while earlier research indicated thattransplant families experience limited type andfrequency of family activities (10). Cohesionamong family members of transplant patientshas been found to be slightly higher than thegeneral population (7); however, more than athird of divorced or separated parents of pediat-ric transplant patients claimed the stress ofraising a chronically ill child contributed tomarital discord (11). Parents of pediatric livertransplant patients also report the stress relatedto ongoing medical care, disruption to familyroutine, and anxiety related to their child�s health(9). The daily adjustments required to accommo-date a child post-transplantation have beenassociated with maternal depression and anxiety(12). Transplantation is far from a panacea, withone parent describing it as a disease just likeESLD that impacts upon family functioning withfinancial burden, adaptation to new parentingroles, and readjustment to family structure (13).Given the previous research detailing alteredfunctioning of families of children with chronicillness or disability (6), the extent to whichfamilies modify or adjust routines to accommo-date children following transplantation warrantsfurther investigation.QOL is now a basic criterion for evaluating the

success of transplant procedures. Similarly toother areas of medicine, costly transplant proce-dures need to be justified in terms of ‘‘QOLgained’’ in addition to ‘‘lives saved.’’ Substantialresearch indicates that liver transplantation isassociated with improved QOL in children withESLD (5, 14, 15); however, there has been littleinvestigation of the way in which pediatrictransplant patients� experiences within the familycontext may relate to QOL. Children�s QOL islikely to be impacted by family routines andactivities, as a child�s understanding of healthand well-being is shaped within the familycontext.The current study sought to investigate family

functioning by exploring the way in whichfamilies modify or adjust routines to accommo-

date children following liver transplantation.The relationship between adjustments to familyroutines and QOL was also investigated. Wehypothesized that families of pediatric livertransplant patients would make more adjust-ments to family routines in comparison withother families in the community and thatincreased adjustments to family routines wouldbe associated with lower QOL among pediatricliver transplant recipients.

Patients and methods

This study was conducted within the RCH Department ofGastroenterology in Melbourne, Australia. Parents of chil-dren aged between three and 16 yr at the time of the studywho received a liver transplant at RCH between 1995 and2010 and were eligible for participation. All children were atleast one-yr post-transplant at the time of study. A com-parative community sample was also recruited. Approvalfor this study was obtained from RCH, RMIT University,and Department of Education and Early ChildhoodDevelopment Human Research Ethics Committees. Writteninformed consent was obtained from parents or caregiversof all children.

Measures

Family Accommodations QuestionnaireThe FAQ (S. Gavidia-Payne, S. Tainsh, unpublished data)was used to measure the extent to which families modify oradjust routines to accommodate their children. The FAQwas developed on the basis of an interview schedule (16)with most previous research being of a qualitative nature.The questionnaire was completed by both transplant andnon-transplant families, with respondents asked to rate theiragreement to each statement on a five-point Likert scale.The FAQ comprises 27 items related to nine areas in whichfamilies may make adjustments to accommodate children(with sample items in parentheses): work (‘‘I need to lookfor jobs that offer flexibility, so that I can look after mychild’’), services (‘‘Our family had to move so that we werecloser to support services for our child’’), the family home(‘‘For the safety and well-being of our child, our familyhome had to be altered’’), domestic workload (‘‘Mydomestic workload is greatly increased, due to my child�sillness’’), childcare tasks (‘‘My child requires constantmonitoring’’), assistance from others (‘‘We ask for assis-tance from our extended family’’), child peer groups (‘‘Ihave to supervise my child while they are playing with otherchildren’’), marital roles (‘‘We often have conflicts and dis-ruption to our marriage due to our child’’), and parentinformation (‘‘We feel that there are professionals we canapproach about our child’’). A total score is also derived,with higher scores indicating more adjustments to familyroutine.

PedsQLTM 3.0 transplant module parent reportThe QOL of transplant recipients was measured using thePedsQLTM 3.0 Transplant Module Parent Report (17).Forty-six items encompass eight scales: barriers to medicalregimen adherence, medication side effects, social relation-ships and transplant, physical discomfort, worries related tohealth status, anxiety regarding medical procedures, impactof transplant on appearance, and communication with

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medical personnel and others about transplant. An overallQOL rating is also derived, with a higher total score indi-cating more optimal QOL.

Data collectionParents of eligible liver transplant patients were identifiedvia their child�s medical records and invited to participate bymail and later sent a questionnaire pack. A follow-up phonecall was made to families who did not return the question-naire within three wk. Data related to patients� age, gender,underlying medical condition precipitating transplantation,time since transplantation, and overnight hospital stayswithin six months prior to the study were also identifiedfrom medical records.A comparative group of families of non-transplant chil-

dren were recruited through local primary and secondaryschools. Children with known chronic illness or medicaldisability were excluded from participation. Study infor-mation was distributed to parents following the approvalfrom school principals, with questionnaires returned viareply-paid postage. Questionnaire return rates for thetransplant and non-transplant groups were 58% and 47%,respectively.

Statistical analysisData were analyzed using SPSS version 19 (18). Two par-ticipants from the transplant group were excluded prior toanalysis because of large amounts of missing questionnairedata. A two-tailed t-test was used to compare the adjust-ments made to family routines by transplant and non-transplant families, and a one-way between-groups manova

was conducted to explore the differences at the subscalelevel. Correlational analyses were performed using Pearsonproduct-moment correlation coefficients to explore theassociation between adjustments to family routines andQOL.

Results

Study population

The final sample for analysis comprised 63families, representing two groups: families ofpediatric liver transplant recipients (n = 30) andfamilies of non-transplant children (n = 33).

Transplant group respondents were mothers(61%), fathers (23%), not specified (10%), or agrandparent (3%). Respondents from thenon-transplant group were mothers (64%) andfathers (36%). The mean age of recipients was9.77 yr (median 10.95 yr), and comprised 52%men and 48% women. Biliary artesia was themost frequent underlying disease precipitatingtransplant (62.2%), while other diagnoses in-cluded metabolic liver diseases such as alpha-1antitrypsin deficiency, OTC, and fulminant liverfailure. Transplant recipients were between 1.1and 12 yr post-transplantation (median 3.4 yr),and the average number of overnight hospitalstays for the transplant group in the six monthsimmediately prior to the study was 1.20(s.d. = 2.27). The mean age of non-transplantchildren was 9.61 yr (median 10 yr), and com-prised 65% men and 35% women. Groups didnot differ statistically in terms of age or gender.

Adjustments to family routines made by transplant and non-transplant families

A significant difference was detected in theadjustments made to family routines by trans-plant and non-transplant families, t(61) = 2.28,p = 0.026. The magnitude of the differencewas moderate (g2 = 0.07). More alterationsto family routines to accommodate childrenwere reported by transplant families (M =68.93, s.d. = 18.25) than non-transplant families(M = 59.15, s.d. = 15.78).Mean scores on the FAQ subscales, summa-

rized in Table 1, indicate that transplant familiesmade more adjustments in all areas except forseeking information related to caring for chil-dren. However, a one-way between-groupsmanova revealed the only difference to reachstatistical significance, using the Bonferroni

Table 1. Two-way multivariate analysis of the functioning of transplant and non-transplant families

Group

Transplant (n = 30) Non-transplant (n = 33)

FAQ subscale Number of items Cronbach�s a df df error F g2 p Value M (s.d.) M (s.d.)

Work 5 0.76 1 61 1.90 0.030 0.173 2.87 (1.04) 2.54 (0.84)Services 3 0.70 1 61 2.26 0.036 0.138 2.52 (1.08) 2.12 (1.04)Home and community 4 0.82 1 61 6.42 0.095 0.014 2.07 (0.94) 1.54 (0.69)Domestic workload 4 0.86 1 61 7.56 0.110 0.008 2.21 (1.08) 1.55 (0.80)Childcare 2 0.60 1 61 8.18 0.118 0.006* 2.63 (1.20) 1.85 (0.98)Assistance 2 0.25 1 61 5.33 0.080 0.024 2.45 (0.90) 1.92 (0.90)Child peer groups 1 – 1 61 4.99 0.076 0.029 2.73 (1.41) 2.03 (1.07)Marital roles 2 0.36 1 61 4.65 0.071 0.035 3.38 (0.80) 2.97 (0.73)Parent information 4 0.67 1 61 3.19 0.050 0.079 2.70 (0.77) 3.05 (0.80)

*Significant at Bonferroni adjusted level of 0.006.

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adjusted alpha level of 0.006, was childcare, F(1,61) = 8.18, p = 0.006; g2 = 0.19. Transplantfamilies reported more adjustments to childcarearrangements than non-transplant families.

Post-transplantation associations between family functioningand QOL

Correlational analyses were used to investigatethe association between adjustments to familyroutines and QOL over time post-transplanta-tion, as measured by the FAQ and PedsQLTransplant Module, respectively. Scores of thePedsQL Transplant Module are summarized inTable 2. No relationship was detected betweentime since transplant and QOL, r = )0.311,n = 30, p = 0.094. A moderate positive corre-lation was found between family functioning andQOL, r = )57, n = 30, p = 0.01, indicatingthat less adjustments to family routines areassociated with QOL improvements. Results alsorevealed that greater time elapsed because trans-plant is associated with fewer adjustments tofamily routines, r = 0.395, n = 30, p = 0.031.Fig. 1 depicts the adjustments made to familyroutines according to time since transplant.

Discussion

While many previous studies have investigatedQOL among pediatric liver transplant recipients,we sought to explore the adjustments made byfamilies to accommodate children who havereceived liver transplants, and the associationbetween these accommodations and the child�sQOL. Results showed that discrepancies in theamount of adjustments made to family routinesby transplant and non-transplant families suggesta need to better understand factors related to thefamily life of transplant recipients and highlightthe importance of promoting post-transplanta-tion family support.

Overall, the results of this study lend support tothe previous findings of transplant families expe-riencing compromised family functioning (10),countering previous claims of no difference in thefunctioning of transplant and non-transplant fam-ilies (7). However, because of the current study�sconceptualization of family functioning as relatedto adjustments made to family routines, suchcomparisons should be interpreted with caution.Parents report most demand on childcare

resources, citing increased need to monitor theirchild and greater need for transportation. Assist-ing parents with childcare difficulties may assistwith the organization of family routines and, inturn, may be associated with positive QOLexperiences for children. Other factors notexplored in this study, such as family structureand resources (e.g., financial), as well as parents�coping strategies, may shed further light on howbest to support families with childcare difficulties.An association was identified between fewer

adjustments to family routines and improvementsto QOL; this may reflect the importance of thehome environment in a child�s recovery followingtransplantation ormay indicate that a better QOLleads to fewer adjustments within the family.Further investigation is required to ascertain thedirection and nature of relationship betweenbetter QOL and adjustments to family routine.Furthermore, the trend of results in this study

indicates that families make fewer adjustments toroutines as time elapses after transplantation.However, the families of patients most yearspost-transplant actually report making moreadjustments in comparison with those whosechild has recently experienced transplantation.

Table 2. PedsQL Transplant Module parent-proxy scores for pediatric livertransplant recipients (n = 30)

Numberof items

Cronbach�sa M (s.d.)

Total score 46 0.93 77.78 (13.45)Barriers to medical regimen adherence 9 0.85 80.19 (18.01)Medication side effects 8 0.54 90.93 (10.67)Social relationships and transplant 8 0.84 72.08 (19.88)Physical discomfort 3 0.71 78.06 (18.63)Worries related to health status 7 0.95 72.73 (28.78)Anxiety regarding medical procedures 4 0.92 67.29 (27.84)Impact of transplant on appearance 3 0.81 80.28 (23.21)Communication with medical personal

and other about transplant4 0.87 74.79 (24.15)

Fig. 1. FAQ scores for pediatric liver transplant patients bytime since transplantation. Higher scores indicate greateramount of adjustment to family routine.

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As our sample is small, this may reflect ananomaly in the experiences of a small number oftransplant families or may be indicative of afamily fatigued after many years of caring for achild with a chronic illness. Further researchregarding this finding is warranted.Several methodological issues limit the con-

clusions that may be drawn from this study.Firstly, this single-center study reflects the uniqueexperiences of liver transplant patients from onelocation. Participants were at different stages ofpost-transplantation, age at transplant was notconsidered, and it was beyond the scope of thisstudy to control for ongoing medical complica-tions that may affect family functioning andQOL. Parental assessment was used in this study;while parental insights into family functioningare appropriate, parents of children withchronic illness tend to over-estimate their child�sdifficulties (5), and transplant children havebeen found to rate their QOL slightly higherthan parents� ratings (17). Further validationof the FAQ measure is also required. Futureresearch should also consider demographic infor-mation, as demographic variables have beenfound to impact family functioning (19). Whilethe response rate was adequate, lack of informa-tion about the families who chose not to partic-ipate may limit generalizability of results.In conclusion, this study offers a new perspec-

tive on the post-transplantation QOL experiencesof children with ESLD by explicit investigationof the way in which family routines are adjustedto accommodate pediatric liver transplant recip-ients. Families� adjustments to accommodatechildren were typified by childcare difficulties,and increased adjustments to family routineswere associated with lower QOL. This studyfurther substantiates the need to assess familywell-being and consider its impact on transplantrecipients.

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