Psychosocial well-being and quality of life in siblings of children with congenital heart disease: A systematic review

Abstract

Congenital heart disease (CHD) is a major global health problem. Until recently, the siblings of this group did not receive much attention. This review, conducted from November 2019 to October 2020, aims to summarize knowledge about psychosocial well-being and quality of life (QoL), associated factors, and interventions for siblings of children with CHD. Systematic searches were conducted in PubMed, PsycINFO, PsycARTICLES, Web of Science via EBSCOhost, and CENTRAL. Twelve articles were included. Results showed that psychosocial well-being was impaired in 14% to 40% of siblings. Negative impact of illness was highest for CHD siblings compared to siblings of children with cancer, cystic fibrosis, or diabetes. QoL was impaired in up to one-third. Siblings of children with CHD and cancer rated their QoL lower than those of siblings of children with cystic fibrosis or type-1 diabetes. Associated factors were sibling age, gender, socioeconomic status, miscarriage, previous sibling death, visibility of illness, and severity of condition. Only one of two interventions focused on siblings of CHD children. Although data are scarce and inhomogeneous, it indicates that siblings of CHD children suffer from lower psychosocial well-being and QoL than siblings of children with other chronic conditions. Interventions to improve their situation should be developed.

Keywords

Heart defects congenital siblings child quality of life

Introduction

Congenital heart disease (CHD) is the result of a defect in the structure of the heart and great vessels of a newborn (Casey, 2016). Almost one-third of all major congenital anomalies consist of heart defects (Dolk et al., 2010). Birth prevalence is estimated at 8–9/1000 live births (Bernier et al., 2010) and has increased substantially over the last decades. Due to massive breakthroughs in cardiac diagnostics and surgery, about 1.35 million newborns are born with CHD worldwide each year (Van der Linde et al., 2011).

Negative consequences of CHD and its treatment—such as intense fear, uncertainty about the future, and the life-threatening potential of the disease—may have profound psychological and social consequences for patients and their families, including siblings, such as loss of social contacts and psychological difficulties (Denniss et al., 2019).

Until recently, siblings of this group have not been a focus of attention. Due to the special needs of children with CHD, siblings are at high risk of being pushed into the background (Naylor and Prescott, 2004). Several studies and meta-analyses suggested that siblings of children with chronic conditions often endorsed more psychosocial difficulties and lower quality of life (QoL) compared to age-matched peers (Sharpe and Rossiter, 2002; Vermaes et al., 2012; Woodgate et al., 2016). They suffer from decreased parental attention and increased parental expectations; partial transfer of responsibility for the child with the chronic condition; conflicts of loyalty, anger, jealousy, or guilt; restriction of societal participation; and stigmatization (Sharpe and Rossiter, 2002; Vermaes et al., 2012; Woodgate et al., 2016). About 30% of siblings of children with chronic conditions are affected by moderate to high stress (Kowalewski et al., 2014).

Recently, Parker et al. (2020) reviewed the literature about the impact of having a sibling with CHD and examined 11 studies. The main conclusions were that siblings experienced adverse life changes that negatively impacted their school performance, behavior, QoL, and health.

To date, no review has investigated the frequency and nature of impairment of psychosocial well-being and QoL within siblings of children with CHD. Furthermore, no reviews have aimed to identify associated factors of these constructs, nor found interventions including these children.

Aims

This systematic review aims to identify the frequency and nature of reduced psychosocial well-being and QoL in siblings of children with CHD to determine if they are affected more strongly and in different ways than siblings of children with other chronic conditions. Moreover, the review seeks to explore factors associated with the effect of a siblings’ CHD as potential starting points for intervention design. Finally, it aims to identify interventions that directly involve siblings of children with CHD and the benefits they provide.

Methods

Data sources, search strategy, and quality appraisal

Methods for this systematic review were prespecified in a protocol that was registered in PROSPERO in November of 2019: https://www.crd.york.ac.uk/prospero/display_record.php?RecordID=156295. A search of PROSPERO and Cochrane databases prior to starting this review found that aside from Parker et al.’s (2020) review, no similar literature reviews were published or were currently being conducted. Based on the key search terms CHD, siblings, psychosocial wellbeing, and QoL that were determined via a PICO scheme, iterative scoping searches were performed that led to the final PubMed search strategy displayed in Table 1.

Table 1.

Literature search strategy.

“Heart Defects, Congenital”[MeSH Terms] OR Congenital Heart Defect*[tiab] OR Heart Abnormalit*[tiab] OR Pediatric Cardiac Surgery*[tiab] OR Congenital Cardiac defect*[tiab] OR Congenital Cardiac diseas*[tiab] OR Child Heart diseas*[tiab] OR Congenital Heart malformation*[tiab] OR Pediatric cardiology[tiab] OR Childhood chronic heart diseas*[tiab]

AND

Siblings [MeSH Terms] OR “Sibling Relations”[MeSH Terms] OR sibling*[tiab] OR sib*[tiab] OR brother*[tiab] OR sister*[tiab]

A broad search strategy was selected to minimize the risk of omitting relevant literature by not matching all search terms reflecting psychosocial well-being and QoL. Systematic searches without publication date limits were performed in PubMed via MEDLINE, PsycINFO, PsycARTICLES, Web of Science via EBSCOhost, and CENTRAL, as well as manual searches of reference lists. Automatic alerts were set up for the searches, which were manually rerun before final analysis on 26 September 2020. Two researchers independently screened articles. Discrepancies were resolved by discussion.

Inclusion and exclusion criteria are displayed in Table 2.

Table 2.

Inclusion and exclusion criteria.

	Included	Excluded
Type of study	Original studies of all designs	Non-original empirical articles
Type of study	Published in peer-reviewed journals	Not published in peer-reviewed journals
Type of participants	Siblings of children with a CHD diagnosis	No self-/other assessment of a sibling of a child with a CHD diagnosis
Type of participants	Parents of a child with CHD and another healthy child
Type of outcome	Psychosocial well-being and QoL	Any outcome not related to psychosocial well-being and QoL or interventions
Type of outcome	Interventions including siblings of children with CHD
Language	English	Others
Language	German	Others

Note: CHD: congenital heart disease; QoL: quality of life.

Instruments measuring psychosocial well-being and quality of life

Instruments measuring impaired psychosocial well-being are differentiated from instruments that claim to measure impaired QoL. This allows distinguishing between a selective influence of a sibling’s CHD on the narrower construct of psychosocial well-being, defined by psychological and social dimensions of QoL (psychological distress, impairment, and behavior problems; Tsang et al., 2012), and an influence on the general QoL, covering psychosocial, physical, and material well-being (Lipscomb et al., 2006).

Results

A total of 1848 references were imported into Covidence (Veritas Health Innovation, 2016). Ultimately, 12 studies were included. Figure 1 depicts the study selection process.

Figure 1.

PRISMA diagram of the study selection process.

The review of the included studies led to the following key themes: (1) psychosocial well-being and subthemes of frequency and nature of impairment, comparisons to siblings of children with other chronic conditions, and comparisons of sibling and parental perspectives; (2) QoL and subthemes of frequency and nature of impairment and comparisons to siblings of children with other chronic conditions; (3) associated factors; and (4) intervention studies.

Description of the chosen studies

All except one study (Wray and Maynard, 2005) revealed incomplete reporting in either methods, results, or discussion. One study reported insufficiently in all areas except the aims (Apley et al., 1967). A table with the results of the quality appraisal can be found under Supplementary Material.

Characteristics of the studies and instruments used are presented in Table 3. The 12 included studies were conducted between 1967 and 2019. Five studies were 24 years or older.

Table 3.

Characteristics of included studies.

Author (date) country	Aim	Design/perspective	Participants	Sample size	Condition	Methods	Results
Apley et al. (1967) UK	To determine the impact of CHD on parents and siblings and how the impact is influenced by cardiac disorder, family characteristics, and medical management	Cross-sectional interview study with unclear quantitative methodology Parental perspective	Mothers of children from Bristol and southwestern England Unclear recruitment procedure designated randomly	Mothers of 70 children with CHD and 155 healthy siblings	Various CHD: VSD, ASD, tetralogy of fallot, patent ductus arteriosus, pulmonary stenosis, coarctation of the aorta, aortic stenosis, and mixed conditions	Psychosocial well-being Sibling reactions: (behavior problems and psycho-behavioral disorders) measured with undefined quantitative surveys completed by researchers, doctors, ward sisters, and school teachers QoL Not measured directly	Psychosocial well-being 27% affected by behavior problems, 13% by psychosomatic disorders, and 24% of both together QoL no results Associated factors Severity of condition: More than twice as many parents of children with incurable CHD reported sibling difficulties, compared to mild CHD. Miscarriage/previous sibling death: 33% of families with sibling difficulties had miscarriages and 18% previous sibling death. In families without sibling difficulties, 4% had previous miscarriages or sibling death
Azhar et al. (2016) Saudi Arabia	To assess the impact of CHD on biopsychosocial aspects of the QoL of patients and their families, including siblings	Cross-sectional interview study and quantitative questionnaires with parents completed by the researcher Parental and sibling perspective	Parents and siblings of children with CHD (58% boys), mean age (SD): 5.65 (4.8) years from one hospital between May 2014 and August 2015	108 parents of children with CHD and 104 healthy siblings	125 (69%) simple CHD, 55 (30%) complex CHD, and 16 (9%) another child affected with CHD (not followed up in hospital)	Psychosocial well-being Not measured QoL The questionnaire developed for the study included the impact of CHD on child, parent, and sibling QoL, as well as family needs and expectations	Psychosocial well-being No results QoL 33% were jealous of their sick sibling. 19% felt neglected by their parents. In 11%, school performance was affected Impact scores (0–100%) of CHD on QoL: siblings’ mean (SD) psychological impact score 24.96 (24.6) versus CHD child: psychological impact score 28.7(28.82) Psychological impact of CHD on siblings almost as high as impact on CHD children Associated factors None
Caris et al. (2018) USA	To assess the impact of HLHS on siblings’ QoL/adjustment and caregivers’ opinion of this effect and to identify factors associated with more negative adjustment in siblings and caregivers	Cross-sectional study and web-based survey distributed via Listservs/social network sites Parental and sibling perspective	Caregivers and siblings of children with HLHS, a very complex CHD, treatable only by three operations Siblings’ mean age (SD): 12.5 (6.3) years. Range 7–30 years (73% of the CHD children had undergone the third operation)	35 caregivers and 32 siblings of 26 families participated = 42 caregiver-sibling pairs (since in nine families, more than one parent and in six families, more than one sibling responded)	HLHS	Psychosocial well-being Two versions of (SPQ; Sahler and Carpenter, 1989) for children and caregivers to assess adjustment of siblings to a child’s chronic illness covering communication, relationships, fear, and contact with other siblings; overall negative adjustment score from 1–4 QoL Not measured with designated questionnaire	Psychosocial well-being Caregivers reported significantly more adjustment problems (M = 2.4, SD = 0.4) than siblings (M = 2.3, SD = 0.3) QoL No results Associated factors Age of siblings: Siblings reported worse adjustment as they grew older; siblings’ age was also associated with caregivers’ adjustment ratings, which decreased as sibling age increased
Havermans et al. (2015) Belgium	To assess self-reported QoL and perception of the impact of chronic illness on siblings	Quasi-randomized controlled trial and quantitative questionnaires Controls: siblings of healthy children Sibling perspective	Siblings of children with four chronic conditions Mean age (SD): CHD 14.3 (2.2), diabetes type-1 13.2 (2.2), cystic fibrosis 13.2 (2.1), and cancer 13.3 (2.5) Control group: 131 siblings, age- and sex-matched	131 siblings of children with CHD 21, diabetes type-1 35, cystic fibrosis 38, and cancer 37	Four chronic medical conditions: cancer, type-1 diabetes, CHD, and cystic fibrosis CHD included tetralogy of fallot (n = 11) and univentricular heart (n = 10). All had at least one major heart surgery	Psychosocial well-being Impact of illness assessed by SPQ (Sahler and Carpenter, 1989), completed at home QoL CHQ-CF87 (Landgraf et al., 1999), control group: CHQ (Landgraf et al., 1999), completed at school	Psychosocial well-being Negative impact of illness was rated highest for CHD siblings, F(4127) = 8.61, p < 0.001 QoL Siblings of children with CHD or cancer rated their QoL lower than the cystic fibrosis or type-1 diabetes group Domains: behavior problems, F(4,127) = 4.03, p < 0.010; mental health, F(4,127) = 4.74, p < 0.010; and self-esteem, F(4,127) = 3.25, p < 0.050 Associated factors None
Janus and Goldberg (1997) Canada	To examine behavior problems of children in families where one child was diagnosed with CHD, in relation to the intensity of treatment required	Cross-sectional study, quantitative, and telephone interviews with mothers and fathers Parental perspective	Parents of children with CHD from 2.5–4 years with a healthy sibling 4–14 years. Siblings mean age (SD): 7.4 (3.3) years	29 children with CHD and 43 healthy siblings	Treatment intensity based on hospitalizations, surgery, current treatment, check-up frequency, and finality of surgical repair	Psychosocial well-being Behavior problems of sick child and sibling: CBCL (Achenbach, 1991) Impact on healthy sibling: parents’ perception of the impact of caring for the CHD child on parental inattention to siblings; “perception of effect on sibling scale” Family life illness accommodation: How often mothers felt they had to take the CHD into account in nine areas of family life QoL Not measured	Psychosocial well-being Occurrence of behavior problems: 14%; not significantly different from the data of a normative sample; 18% (Hobbs et al., 1985) QoL No results Associated factors Severity of condition: Siblings had more behavior problems (mother’s reports) when the patient’s condition required less treatment Stronger impact on sibling when treatment was more intensive Family life illness accommodation: higher in families of siblings with behavior problems
Lavigne and Ryan (1979) USA	To compare the psychological adjustment of siblings of children with chronic illnesses or handicaps and siblings of healthy children	Cross-sectional study with a healthy control group and quantitative questionnaires Parental perspective	Parents of siblings of children with three chronic medical conditions, recruited from hospitals Healthy controls recruited from a school. Data completed on oldest and youngest siblings age 3–13	177 mothers, 20 fathers, and 6 stepparents/guardians of siblings of children with CHD (n = 57), hematology conditions (n = 62), plastic surgery (n = 37), and healthy children (n = 46)	CHD: various cardiac conditions, the largest group, VSD (n = 11) Hematology: all, but two had leukemia or cancer (n = 23) Plastic surgery: various diagnoses, largest group cleft palate, cleft lip, and palate (n = 12)	Psychosocial well-being Behavior problems: LBCL (behaviors which reflect adjustment problems, Miller, 1977) QoL Not measured directly	Psychosocial well-being Social withdrawal, overall disturbance, and irritability were worse for illness groups than for controls QoL No results Associated factors Severity of condition: no relationship between severity of illness and psychopathology within the CHD group Visibility of handicap: siblings of patients with visible handicaps (plastic surgery) were significantly more socially withdrawn than siblings of patients with invisible illnesses (CHD and hematology), F(1191) = 14.98, p < 0.010
Menke (1987) USA	To explore the impact of a child’s chronic illness on school-aged siblings in the family system	Qualitative interview Study Sibling perspective	Siblings of children with chronic medical conditions 6–12 years (mean age of 9.6 years)	72 siblings, 39 girls, and 33 boys from 53 families. Cancer (n = 20), cystic fibrosis (n = 15), CHD (n = 14), myelomeningocele (n = 12), and severe burns (n = 11)	See column sample size. Conditions not further specified	Psychosocial well-being Structured interview developed by the researcher, 45 min, 90% in participants’ home (full interview provided) QoL Not measured	Psychosocial well-being Worries about self, sibling, and parents/family: the CHD group more likely to have concerns about the ill child (57%) compared to children with burns (36%) and cancer (20%) QoL No results Associated factors Age of sibling/position in sibling order: More siblings older than the sick child had protective concerns
Mughal et al. (2011) Pakistan	To assess the socioeconomic status, treatment, and socioeconomic impact of a child’s CHD on families	Interview study with parents before and after treatment and at the time of the child’s discharge from the hospital Parental perspective	Parents of children undergoing either cardiac surgery or angiography (mean age of patients: 3.3 years)	Parents of 211 children (60% male) with cardiac surgery (n = 164) versus angiographic cardiac intervention (n = 47)	CHD: various cardiac conditions	Psychosocial well-being Not measured directly QoL Schooling and health of siblings	Psychosocial well-being No results QoL Negative effect on schooling (23%) and health of siblings (26%) Associated factors None
Redshaw and Wilson (2012) Australia	To analyze statements made by parents in interviews regarding an evaluation of the Heart Beads Program which commented on benefits for siblings	Qualitative interview study Parental and sibling perspective	Families of children with CHD and a healthy sibling who participated in the Heart Beads Program	Analyses focused on 10 of 19 interviews in which siblings were mentioned	CHD:no definition provided	Intervention CHD children collect beads for each intervention. Qualitative interviews regarding an evaluation of the Heart Beads Program (example questions provided)	Intervention Touching and explaining beads: helped parents to explain what was happening (providing information and explaining) Collecting beads to include a sibling: letting sibling thread beads, as a way of including the sibling in the process of meaning construction around the sibling’s CHD. Parents state the program enabled siblings to feel included
Van der Mheen et al. (2019) Netherlands	To determine the effectiveness of an innovative psychosocial intervention, named CHIP-Family, to improve the psychosocial well-being of children with CHD and their families	Single-blinded parallel randomized controlled trial, quantitative questionnaires, and intervention study Parental perspective	Children with CHD and their families (parents and siblings) participated in the workshop Parents and CHD child completed questionnaires	93 CHD children, mean age (SD): 5.34 (1.27) years, randomized to CHIP-Family (n = 49), or care-as-usual (i.e., no psychosocial care; n = 44)	Families of children who underwent at least one invasive medical procedure for CHD (e.g., cardiac catheterization and/or open-heart surgery)	Intervention CHIP-Family: 1-day group workshop for parents, children, and siblings Areas covered for children/siblings: exercises for strengthening self-esteem, regulating emotions, relaxation, problem-solving skills, positive thinking, and sports Family functioning, the only measure including siblings, was assessed through the general functioning subscale of the Family Assessment Device (12 items; Epstein et al., 1983)	Intervention Family functioning: no difference between the CHIP-Family and care-as-usual group No direct effect measures for siblings, since the intervention was not aimed directly at siblings, but each CHD child was allowed to bring a 4- to 10-year-old sibling/friend to normalize participation and to stimulate practice at home
Williams et al. (1993) Philippines	To explore the effects of pediatric chronic illness on sibling and maternal activity	Cross-sectional study, qualitative interviews, and quantitative questionnaires Parental perspective	100 families of children with neurological and cardiac conditions. Families primarily of lower socioeconomic status, with 4–6 children. Siblings were between 6 and 18 years old and emotionally and physically healthy	57 mothers of children with CHD or acquired cardiac disease 43 mothers of children with neurological diseases	Either CHD or acquired cardiac disease and neurological diseases	Psychosocial well-being Structured interviews/45 min, partly based on Duvall’s (1977) concept of family development tasks (sample items provided) QoL Not measured directly	Psychosocial well-being Extent of decrease in caretaking activities of siblings was significantly greater among mothers of children with CHD than those with neurologic conditions, t(98) = 2.74, p < 0.010 QoL No results Associated factors Socioeconomic status: Most families were of low socioeconomic status. Mothers reported significant increase in siblings’ household, t(98) = 2.32, p < 0.050, and decrease in social activities, t(98) = 5.39, p < 0.010 Female gender: Female siblings were given twice as many caretaking activities as male siblings
Wray and Maynard (2005) UK	To assess maternal perceptions of the impact of CHD on the child, parents, and siblings, and to determine whether there were differences between diagnostic groups, or those with and without other health problems	Cross-sectional mixed-methods study and postal questionnaires with quantitative and qualitative questions Parental perspective	Parents of siblings of children with CHD that had been inpatients on one cardiology ward between 1995 and 1999	209 parents of siblings of children with CHD	The majority had acyanotic or cyanotic lesions, 24 had transplantation, 11 had miscellaneous CHD, e.g., rheumatic valvular disease, cardiomyopathy, arrhythmias, or Kawasaki disease	Psychosocial well-being Questionnaires developed by researchers, measuring the impact of CHD on activities, family relationships, care issues, and education QoL Not measured directly	Psychosocial well-being In 30% of the families, siblings were perceived to be negatively affected by CHD by extra attention to sick child; prevented from doing things as a family; fear of getting too close to sick sibling; feeling that sick child does not have same rules to adhere to; feeling left out; anxiety/depression; anger; intolerance; jealousy; resentment; and insecurity QoL No results Associated factors Severity of condition: 60% of siblings of transplanted children, 43% of siblings of cyanotic children, and 16% of siblings of acyanotic children affected and 25% of parents gave more time to the ill child

Note: CHD: congenital heart disease; QOL: quality of life; VSD: ventricular septal defect; ASD: atrial septal defect; HLHS: hypoplastic left heart syndrome; SPQ: Sibling Perception Questionnaire; CBCL: Child Behavior Checklist; LBCL: Louisville Behavior Checklist; CHIP: Congenital Heart Disease Intervention Program.

Three studies were conducted in the United States, two in the United Kingdom, and one each in Australia, Belgium, Canada, Netherlands, Pakistan, Philippines, and Saudi Arabia.

Two studies used both quantitative and qualitative methods (Williams et al., 1993; Wray and Maynard, 2005), eight studies used quantitative questionnaires (Apley et al., 1967; Azhar et al., 2016; Caris et al., 2018; Havermans et al., 2015; Janus and Goldberg, 1997; Lavigne and Ryan, 1979; Mughal et al., 2011; Van der Mheen et al., 2019), and two studies applied qualitative methodology (Menke, 1987; Redshaw and Wilson, 2012).

Two studies used sibling reports only (Havermans et al., 2015; Menke, 1987), seven studies focused on the proxy reports of parents exclusively (Apley et al., 1967; Janus and Goldberg, 1997; Lavigne and Ryan, 1979; Mughal et al., 2011; Van der Mheen et al., 2019; Williams et al., 1993; Wray and Maynard, 2005), and the remaining three studies reported both sibling and parent perspectives (Azhar et al., 2016; Caris et al., 2018; Redshaw and Wilson, 2012).

Four studies investigated siblings of children with CHD and other chronic conditions (Havermans et al., 2015; Lavigne and Ryan, 1979; Menke, 1987; Williams et al., 1993). Two studies provided a control group of siblings of healthy children (Havermans et al., 2015; Lavigne and Ryan, 1979).

Reported information and sample sizes varied widely between studies. Some reported numbers of included families without indicating the number of siblings, some reported the number of siblings, and some reported the numbers of children with CHD (Apley et al., 1967; Azhar et al., 2016; Caris et al., 2018; Janus and Goldberg, 1997).

Most studies included siblings of children with various types of CHD (Apley et al., 1967; Azhar et al., 2016; Havermans et al., 2015; Lavigne and Ryan, 1979). A single study (Caris et al., 2018) exclusively considered children with hypoplastic left heart syndrome (HLHS), one of the severest forms of CHD. Another study did not further define the participants’ CHD (Menke, 1987).

Seven studies provided information about psychosocial well-being (Apley et al., 1967; Caris et al., 2018; Janus and Goldberg, 1997; Lavigne and Ryan, 1979; Menke, 1987; Van der Mheen et al., 2019; Williams et al., 1993; Wray and Maynard, 2005). One study directly investigated QoL with a designated questionnaire (Azhar et al., 2016), while one study reported aspects of QoL (Mughal et al., 2011). Another study reported both psychosocial well-being and QoL (Havermans et al., 2015).

A total of seven studies reported factors associated with psychosocial well-being (Apley et al., 1967; Caris et al., 2018; Janus and Goldberg, 1997; Lavigne and Ryan, 1979; Menke, 1987; Van der Mheen et al., 2019; Williams et al., 1993).

Two intervention studies were identified; one directly included siblings (Redshaw and Wilson, 2012; Van der Mheen et al., 2019).

Psychosocial well-being

Frequency and nature of impairment of psychosocial well-being

A UK study detected impaired psychosocial well-being in up to 40% of siblings. Further, 27% were affected by behavior problems, 13% by psychosomatic disorders, and 24% by both (Apley et al., 1967). However, the study had considerable methodological weaknesses including an unclear recruitment procedure and undefined questionnaires; it also was conducted more than 50 years ago. Another UK study (Wray and Maynard, 2005) found that 25% of parents gave more time to the sick child, and 30% of siblings felt impaired by the extra attention to the sick child, felt they were being prevented from doing things as a family, and indicated fear of getting too close to the sick sibling. Canadian researchers Janus and Goldberg (1997) reported that clinical behavior problems were prevalent in 14% of children, which was not significantly different from the data of a normative sample (Hobbs et al., 1985).

Psychosocial well-being of CHD siblings compared to other illness groups

Belgian researchers examined siblings of children with cancer, type-1 diabetes, cystic fibrosis, CHD, and a control group. Negative impact of illness was rated highest for CHD siblings, F(4127) = 8.61, p < 0.001 (Havermans et al., 2015). A Philippine study compared reports of mothers of children with cardiac and neurological conditions. The extent of decrease in caretaking activities of siblings was significantly greater among mothers of children with CHD than those with neurologic conditions, t(98) = 2.74, p < 0.010 (Williams et al., 1993). Another study with siblings of children with cancer, cystic fibrosis, CHD, myelomeningocele, and severe burns showed that CHD siblings were more likely to have concerns about the CHD children (64%), compared to children with burns (36%) and cancer (20%) (Menke, 1987).

Psychosocial well-being: Sibling versus parent perspective

A US study investigated the degree of adjustment of siblings to their brothers and sisters’ HLHS (Caris et al., 2018) and found that caregivers reported significantly more adjustment problems (M = 2.4, SD = 0.4) than siblings themselves (M = 2.3, SD = 0.3; overall negative adjustment score: 1–4, higher scores = more negative adjustment).

Factors associated with psychosocial well-being

One study found that siblings’ reported adjustment grew worse with age and that increasing sibling age was associated with caregivers’ negative adjustment ratings (Caris et al., 2018). Another study found that typically siblings who were older than the child with the chronic condition had protective concerns, albeit for all five disease groups (see line seven in Table 3) (χ² = 7.27, df = 4, p = 0.010; Menke, 1987).

Williams et al.’s (1993) study revealed that female siblings were given twice as many caretaking activities as their male counterparts. The families participating in the study were primarily of lower socioeconomic status and typically had four to six children. Mothers reported a significant increase in sibling’s household help (mean difference –0.5, t(98) = 2.32, p < 0.050) and a decrease in social activities (mean difference 0.99, t(98) = 5.39, p < 0.010).

One study found that 33% of siblings classified as maladjusted came from families with previous miscarriages and 18% from families with previous sibling death (Apley et al., 1967). These characteristics applied only to the 4% of siblings not classified as maladjusted.

Siblings of patients with visible conditions (i.e., plastic surgery) were significantly more socially withdrawn than siblings of patients with invisible conditions such as CHD/hematology, F(1191) = 14.98, p < 0.010 (Lavigne and Ryan, 1979).

Results concerning severity of condition were inconsistent. Within one study, more than twice as many parents of the most severely ill children (incurable CHD) reported sibling difficulties, compared to siblings of the least severely ill children (Apley et al., 1967). Another study found siblings of children with less complex CHD were affected negatively in 16% of families, compared to 43% of siblings of children with complex CHD, and 60% of children with transplants (Wray and Maynard, 2005). Another study revealed that parents perceived themselves as having increased inattention toward siblings (Janus and Goldberg, 1997) when the ill child’s treatment was more intensive, whereas siblings had more behavior problems when the patients’ condition required less treatment (Janus and Goldberg, 1997). Greater illness accommodation, defined as how often mothers had to take the patients’ condition into account, was reported in families with siblings with behavior problems. In contrast, a US study (Lavigne and Ryan, 1979) failed to find a relationship between severity of illness and psychopathology in CHD siblings.

Quality of life

Frequency and nature of impairment of QoL

A study from Saudi Arabia (Azhar et al., 2016) found that QoL was affected in up to one-third of siblings. Specifically, 33% were jealous, 19% felt neglected by their parents, and 11% had impaired school performance. Psychological impact on the siblings’ QoL (M = 24.96, SD = 24.6) was almost as high as the impact on CHD children (M = 28.7, SD = 28.82; impact scores: 0–100; Azhar et al., 2016). A Pakistani study revealed a negative effect on schooling in 23% and health in 26% of siblings (Mughal et al., 2011).

Quality of life of CHD siblings compared to other illness groups

A Belgian study (Havermans et al., 2015) revealed that siblings of children with CHD or cancer rated their QoL lower than those of children with cystic fibrosis or type-1 diabetes regarding behavior and internalizing problems, F(4127) = 4.03, p < 0.010; mental health, F(4127) = 4.74, p < 0.010; and self-esteem, F(4127) = 3.25, p < 0.050.

Interventions

Two intervention studies were identified. An Australian qualitative interview study (Redshaw and Wilson, 2012) did not target siblings directly but provided benefits to them through the Heart Beads Program. Children received beads for each intervention, such as conversing with their siblings about their feelings and experiences related to CHD. Parents emphasized the benefits of the beads. For instance, touching beads enabled parents to explain what was happening, and collecting and threading beads helped siblings in the process of meaning construction around CHD.

The Congenital Heart Disease Intervention Program (CHIP)-Family was the first intervention that directly included siblings (Van der Mheen et al., 2019). The program consisted of a 1-day group workshop and aimed to improve the psychosocial well-being of children with CHD, their siblings, and parents. Children performed exercises for self-esteem, emotion regulation, relaxation, problem solving, positive thinking, and physical training. Parents underwent problem prevention therapy, psychoeducation, parenting training, and medical education. The study contained no sibling questionnaire; “Family Functioning” was the only survey including siblings. No significant differences were found between the CHIP-Family and care-as-usual groups (p = 0.628, t-values not reported).

Discussion

The present review, in addition to Parker et al.’s (2020), provides insight into the frequency and nature of impairment of psychosocial well-being and QoL in siblings of children with CHD. The study aimed to determine associated factors of these constructs and identified one intervention directed at them. Although the review was limited by heterogeneous databases and by studies having different objectives, samples, instruments, and study quality, some valuable information was extracted.

Psychosocial well-being and quality of life

Psychosocial well-being was impaired in 14%–40% of siblings, which implies that the psychosocial well-being of at least 60% remained unaffected (Apley et al., 1967; Janus and Goldberg, 1997; Wray and Maynard, 2005). Methodological weaknesses such as unclear recruitment procedures, undefined questionnaires, and the age of Apley et al.’s (1967) study render it incomparable to contemporary clinical care. Although its results must be interpreted with caution, this seminal study was the first to address the difficulties of siblings of children with CHD. Janus and Goldberg (1997), who found that 14% of siblings were affected, only considered participants with problems within a clinical range (1 SD above average). Thus, the actual proportion of those affected in their sample was likely higher.

Quality of life was impaired in up to one-third of siblings, which similarly suggests that the QoL of at least two-thirds was unimpaired (Azhar et al., 2016; Mughal et al., 2011). Psychosocial well-being was affected negatively in a higher percentage of CHD siblings than QoL, which suggests that factors already covered by QoL questionnaires, such as strong school bonding or close relationships, act as protective factors that reduce psychosocial stress (Werner, 2013).

One study found that the overall negative impact of illness on psychosocial well-being was rated highest for CHD siblings, and siblings of children with CHD or cancer rated their QoL lower than those of children with cystic fibrosis or type-1 diabetes (Havermans et al., 2015). An explanation might be the permanently life-threatening potential of CHD, which results in elevated levels of fear and hospital stays with absent parents from the moment of the sibling’s birth. This typically does not occur with children with cancer, cystic fibrosis, or diabetes. The difference is especially obvious with complex CHD, which accounts for about 40% of those affected, as lifelong recurring medical intervention is required with no chance of recovery (Caris et al., 2018). CHD siblings also were more likely to have concerns about the CHD child relative to siblings of children with burns and cancer (Menke, 1987).

The study by Azhar et al. (2016) found the psychological impact of CHD on the siblings’ QoL was almost as high as the impact on affected children. This may be because siblings often suffer from feelings of fear and uncertainty and must cope with the physical absence of at least one parent during hospital stays. Additionally, enduring parental distress can lead to refusal of parental attention and affection (Caris et al., 2018).

Factors associated with psychosocial well-being

In one study, siblings and parents reported worse adjustment as sibling age increased, although the study was restricted to children diagnosed with HLHS, one of the severest forms of CHD (Caris et al., 2018). Another study found that siblings older than the CHD child had more protective concerns about them (Menke, 1987). With increasing age, siblings could develop a better understanding of the consequences of their brothers or sisters' CHD. As siblings age, their cumulative number of stressful experiences increases, such as parental absence due to the CHD child’s hospital stays. Although results suggested that increasing age was a risk factor, the data are too scarce to draw final conclusions.

One study showed that female siblings assumed twice as many caretaking activities (Williams et al., 1993), implying that female gender negatively influenced free time. However, the findings were not restricted to CHD siblings, and the affected families were primarily of lower socioeconomic status, with about four to six children. The results might therefore not apply to siblings of families with fewer children and higher socioeconomic status (Williams et al., 1993). Additionally, the study was conducted in the Philippines more than 25 years ago, where a traditional image of women still prevailed (Lauser, 2008). It is questionable whether a similar finding would occur today in a society with a less traditional image of women.

Williams et al.’s (1993) results concerning a significant increase in siblings’ household activities and a decrease in social activities affirm the negative influence of a family’s lower socioeconomic status. Financial difficulties might lead to a higher general stress level within a family because financial resources to mitigate the adverse effects of a chronic condition are lacking. Additionally, the extent of decrease in sibling caretaking was significantly greater among mothers of children with CHD than those with neurological conditions (Williams et al., 1993). This might be a result of the frequent feeding difficulties of CHD children (Clemente et al., 2001).

Previous miscarriage or death of a sibling was suggested by one study to negatively influence the psychosocial well-being of siblings (Apley et al., 1967). Whether the previous experience of losing sibling functions as a type of negative priming needs further investigation (Kempson et al., 2008).

One study compared psychological adjustment of siblings of children with CHD, hematology, plastic surgery conditions, and healthy children (Lavigne and Ryan, 1979). No relationship between severity of illness and psychopathology was found within the CHD group (Lavigne and Ryan, 1979). Moreover, siblings of children with visible plastic surgery conditions were significantly more socially withdrawn than siblings of children with invisible conditions such as CHD, which suggests the invisibility of a condition may be a potential protective factor (Lavigne and Ryan, 1979). Nevertheless, the study by Lavigne and Ryan (1979) was conducted over 40 years ago when plastic surgery techniques to treat conditions like cleft palate were still unsatisfactory and higher levels of stress from such a condition were more likely than today. Also, CHD cannot be called invisible per se. Especially in complex CHD, cyanosis and the resulting blue skin color, as well as a highly perceptible limitation of physical exercise capacity and operation scars, are prevalent (Feltez et al., 2015; Horner et al., 2000).

Regarding severity of condition, ambiguous results were found. Wray and Maynard (2005) found siblings of children with mild CHD were negatively affected about three times less than siblings of children with complex CHD and four times less than siblings of children with transplants, leading to the conclusion that severity of CHD is positively correlated with negative effects. Another study discovered twice as many parents of children with incurable CHD reported sibling difficulties compared to those with mild CHD (Apley et al., 1967). Other researchers failed to find a relationship between severity of illness and psychopathology within the CHD group (Lavigne and Ryan, 1979). One study found that parents experienced siblings as having more behavior problems when the condition required less treatment, although stronger parental inattention toward siblings was perceived by parents when treatment was more intensive (Janus and Goldberg, 1997). These findings indicated that siblings were at the highest risk for behavior problems either in families with children with the most complex CHD or in families characterized by a high level of family accommodation and a relatively mild condition, which may lead to siblings’ poor acceptance of the situation (Janus and Goldberg, 1997). Another potential explanation was siblings’ greater striving for normality when the condition was relatively mild, which might be perceived as unreachable and thereby result in elevated levels of frustration (Janus and Goldberg, 1997).

Due to the studies’ restricted statistical analyses, causal conclusions regarding risk factors cannot be drawn. Additionally, there is a lack of results concerning protective factors, as is found in the context of research on resilience, such as low impulsivity, close relationship to at least one parent, intensive parental supervision, sound academic achievement, strong school bonding, a positive class climate, and living in a nondeprived neighborhood (Werner, 2013). Since up to two-thirds of siblings show no impairment in either psychosocial well-being or QoL, it is probable that protective factors function within these families. To better understand which factors influence psychosocial well-being and QoL of siblings, and in which direction, future research should consider protective factors as well as risk factors and use statistical methods that allow causal conclusions to be drawn, such as regression analyses, analyses of variance, and path models. Before-and-after designs are difficult to implement in this field because CHD cannot be predicted until shortly before birth, and it is therefore impossible to test siblings beforehand (Friedberg et al., 2009).

Interventions

In Redshaw and Wilson’s (2012) study of the Heart Beads Program, parents reported a positive effect on their healthy siblings by involving them in the creation of a family narrative of their siblings’ CHD and by showing appreciation of their feelings and experiences. This enabled siblings to play an active role in the family’s coping and reduced behavior problems. The study of the CHIP-Family program (Van der Mheen et al., 2019), which was the first intervention directed at siblings of CHD children, similarly provided information and explanations about siblings’ experiences. The positive coping mechanisms introduced by the CHIP workshops might have helped the whole family to integrate the child’s CHD into their daily life, although the results did not show significant differences between the intervention and care-as-usual groups (Van der Mheen et al., 2019). Inclusion of a questionnaire for siblings would have provided better insight into a possible improvement of their psychosocial well-being through the intervention.

Clearly, more interventions must be developed, and studies will have to determine their effects, the mechanisms of these effects, and predictors of benefits.

Strengths

To the best of our knowledge, this is the first systematic review that comprehensively covers how the frequency and nature of a siblings’ CHD affects their healthy sibling’s psychosocial well-being and QoL, factors associated with this influence, and intervention studies directed at these children.

Another strength is this review’s quality standard, which meets the highest methodological requirements in the areas of review protocol, study selection, data collection, and risk of bias assessment. The AXIS tool (Downes et al., 2016) was chosen for quality appraisal, which does not provide a numerical scale to assess study quality. Numerical scales can be problematic as their outputs are not linear and as such difficult to sum or weight (Greenland and O’Rourke, 2001), especially when studies are inconsistent in design and outcome variables like the included studies.

Limitations

Limitations include inconsistencies in the reviewed studies’ design and outcome variables, which complicate comparability and render meta-analysis impossible.

The transferability to current siblings of CHD children is questionable due to the high age (>24 years) of five of the included studies (Apley et al., 1967; Janus and Goldberg, 1997; Lavigne and Ryan, 1979; Menke, 1987; Williams et al., 1993) and considering the highly improved treatment options and changed family and environmental factors, such as the necessity of employment for both parents.

Differences between the health systems of the nine study countries further limit the generalizability of the results. Congenital heart disease has a different social and financial impact on families and siblings in countries with state insurance and easy access to maximum medical care, such as the Netherlands (Van de Ven and Schut, 2008), than in countries where only a few, wealthy people have access to health care, such as Pakistan (Kurji et al., 2016).

Future research

Qualitative interview studies were helpful to identify the needs of CHD siblings. Based on their results and the results of our review, controlled cross-sectional studies comparing contemporary parental and sibling reports are required to better understand the influence CHD has on siblings’ psychosocial well-being and QoL. By emphasizing the sibling perspective, eventual parental overestimation (Caris et al., 2018), as well as potential sibling underestimation, could be avoided.

To empower the development of a tailored intervention directed at CHD siblings, it is also crucial to precisely estimate who is at risk. Possible risk factors, such as sibling age, gender, socioeconomic status, miscarriage or previous sibling death, and severity of condition should be more deeply investigated, as well as intra- and interpersonal protective factors like low impulsivity, close relationships, or strong school bonding. Siblings were most stressed by mental health problems—such as anxiety, sadness, depression, worries, jealousy, and feelings of rejection—and behavior and school problems. Exercises for these domains could represent starting points for an intervention.

Trans-diagnostic aspects of research on siblings of children with a chronic condition, which were identified by a systematic review on siblings of youth with developmental disabilities, should be considered (Tudor and Lerner, 2015). These include the need for an effective assessment of sibling needs and the benefits of psychoeducation, identification of the family system factors, the need for manualized treatment programs, and collaborative approaches to performing sibling interventions.

Implications for practice

Healthcare providers and social work professionals should take the above results into account when dealing with siblings of children with CHD and their families. They should remind caregivers of the emotional challenges siblings face to prevent them from becoming so-called shadow children, and they should learn from families that appear better adapted to the adverse conditions. For instance, these actions could be realized in self-help groups.

Conclusion

Psychosocial well-being and QoL were impaired in more than one-third of siblings. The negative impact of illness was highest for CHD siblings, and they rated their QoL lower relative to siblings of children with other chronic conditions. Data are inconclusive regarding associated factors. Future research should focus on these points to support the development of interventions to improve CHD siblings’ situations. Practitioners, as well as parents, should incorporate the above results when dealing with CHD siblings.

Supplemental Material

sj-pdf-1-chc-10.1177_13674935211012933 –x2013; Supplemental Material for Psychosocial well-being and quality of life in siblings of children with congenital heart disease: A systematic review

Supplemental Material, sj-pdf-1-chc-10.1177_13674935211012933 for Psychosocial well-being and quality of life in siblings of children with congenital heart disease: A systematic review by Alice S Schamong, Hannah Liebermann-Jordanidis, Konrad Brockmeier, Elisabeth Sticker and Elke Kalbe in Journal of Child Health Care

Footnotes

Declaration of conflicting interests

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Funding

The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was financially supported by “Verein der Freunde und Förderer des Herzzentrums des Universittsklinikums Köln e.V.” for this article.

ORCID iD

Alice S Schamong

Supplemental material

Supplemental material for this article is available online.

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