PediatricCardiologySystematicReview2015

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Familial Impact and Coping with Child Heart Disease: A Systematic Review
Article in Pediatric Cardiology · January 2015
DOI: 10.1007/s00246-015-1121-9 · Source: PubMed
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Familial Impact and Coping with Child
Heart Disease: A Systematic Review
Alun C. Jackson, Erica Frydenberg,
Rachel P.-T. Liang, Rosemary O. Higgins
& Barbara M. Murphy
Pediatric Cardiology
ISSN 0172-0643
Pediatr Cardiol
DOI 10.1007/s00246-015-1121-9
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Pediatr Cardiol
DOI 10.1007/s00246-015-1121-9
REVIEW ARTICLE
Familial Impact and Coping with Child Heart Disease:
A Systematic Review
Alun C. Jackson • Erica Frydenberg •
Rachel P.-T. Liang • Rosemary O. Higgins
Barbara M. Murphy
•
Received: 13 July 2014 / Accepted: 16 January 2015
Ó Springer Science+Business Media New York 2015
A. C. Jackson (&) R. P.-T. Liang R. O. Higgins B. M. Murphy
Heart Research Centre, 14-20 Blackwood Street,
North Melbourne, VIC 3051, Australia
e-mail: aluncj@unimelb.edu.au
enhance family coping. Twenty-five studies were selected
for inclusion, using the PRISMA guidelines. Thematic
analysis identified a number of themes including psychological distress and well-being, gender differences in
parental coping, and variable parenting practices and a
number of subthemes. There is general agreement in the
literature that families who have fewer psychosocial
resources and lower levels of support may be at risk of
higher psychological distress and lower well-being over
time, for both parent and the child. Moreover, familial
factors such as cohesiveness and adaptive parental coping
strategies are necessary for successful parental adaptation
to CHD in their child. The experiences, needs and ways of
coping in families of children with CHD are diverse and
multi-faceted. A holistic approach to early psychosocial
intervention should target improved adaptive coping and
enhanced productive parenting practices in this population.
This should lay a strong foundation for these families to
successfully cope with future uncertainties and challenges
at various phases in the trajectory of the child’s condition.
A. C. Jackson E. Frydenberg R. P.-T. Liang
Melbourne Graduate School of Education, The University of
Melbourne, Parkville, VIC 3010, Australia
Keywords Child heart disease Familial impact Parenting Coping Social support
A. C. Jackson
Centre on Behavioural Health, University of Hong Kong, 2/F
The Hong Kong Jockey Club Building for Interdisciplinary
Research, 5 Sassoon Road, Pokfulam, Hong Kong
Introduction
Abstract Families of children with congenital heart disease (CHD) cope differently depending on individual and
familial factors beyond the severity of the child’s condition. Recent research has shifted from an emphasis on the
psychopathology of family functioning to a focus on the
resilience of families in coping with the challenges presented by a young child’s condition. The increasing number of studies on the relationship between psychological
adaptation, parental coping and parenting practices and
quality of life in families of children with CHD necessitates
an in-depth re-exploration. The present study reviews
published literature in this area over the past 25 years to
generate evidence to inform clinical practice, particularly
to better target parent and family interventions designed to
R. O. Higgins
Department of Physiotherapy, Faculty of Medicine, Dentistry
and Health Sciences, The University of Melbourne, Parkville,
VIC 3010, Australia
B. M. Murphy
Faculty of Medicine, School of Psychological Sciences,
Dentistry and Health Sciences, The University of Melbourne,
Parkville, VIC 3010, Australia
Several studies have reported that the presence of chronic
conditions in children places the well-being of the entire
family at risk as the burden of care increases vulnerability
to major psychological and social disturbances [25, 40, 61].
Diagnosis is recognised as a particular point of vulnerability [54, 56]. Graetz et al. [20] note the range of new
familial adjustment tasks post-diagnosis, whilst others [51]
go further in identifying diagnosis, the commencement of
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treatment, transition from hospital to home and relapse as
all being potential points of elevated stress, even in families who are otherwise coping well, which is the majority
of families [50].
Two decades of research into family coping with
childhood illness, including behavioural issues [16], has
seen a welcome shift away from the psychopathology of
family functioning to a focus on family resilience in coping
with the challenges associated with their child’s illness [30,
34]. Coping has been defined [35] as the ability to understand the meaning of the illness and its treatment; manage
emotional reactions and continue to undertake necessary
actions; fulfil responsibilities; and provide support. Coping
also refers to the process of managing stressful demands
[23]. The majority of studies on families’ response to
childhood conditions suggest that overall, most families
adjust well [31, 35, 36, 51]. However, a significant subset
of parents of children with CHD (PCCHD) has difficulty
coping in the short and long term [21, 35, 36]. Parental
coping has been demonstrated to be a significant predictor
of coping and adjustment in children with conditions
including epilepsy [47], sickle cell disease [59] and
childhood leukaemia [35, 36, 48]. PCCHD are not always
able to make the best use of informational, practical and
emotional support provided at the point of diagnosis or at
the site of ongoing treatment. They often express a need for
time and space to process emotions such as grief, fear and
loss and to assimilate the new situation into their repertoire
of existing coping patterns as well as learning enhanced
coping skills [26–28]. These emotional, well-being and
coping enhancement needs have been recognised as relevant to child heart disease families at various phases of the
child’s illness trajectory [22, 52, 58]. For example, acute
stress disorder in parents after their infant’s cardiac surgery
has been identified [15], as have issues in paternal attachment to their infant at the time of surgery [4].
The purpose of this systematic review was to examine
evidence of how child heart disease families are impacted
by the challenges presented by a young child’s condition,
in order to better target parent and family interventions
designed to enhance the coping capacity of these families.
Method
Procedure
Multi-database Boolean searches were conducted in EBSCOhost, CSA Illumina and JSTOR using the search term
‘family coping’ AND ‘child heart disease’. The database
search was limited to English-language scholarly articles of
peer-reviewed journals published from January 1990
onwards. The top 225 results were selected by relevance
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from the 53,506 populated results and imported into EndNote X7. An initial sort of the titles and abstracts resulted
in 82 articles being rejected as either they were duplicates
or there was no full-text report available. An additional 42
studies were identified from manual searches of the reference lists of key studies identified. The resulting 185 articles were subjected to detailed assessment based on the
following inclusion and exclusion criteria:
Sample
Studies were considered for the current review if they met
the following inclusion criteria:
1.
2.
3.
4.
5.
The study sample comprised families with dependent
children aged under 21 years with clearly identified
congenital heart disease (CHD) (not acquired)
No comorbidity or other presenting chronic illness in
the study group although these may be present in
control groups
Studies focused on the familial impact of child heart
disease and family coping and impact on, and coping
by, parents of children with CHD (PCCHD)
Explicit methodological design (qualitative, quantitative and/or observational) with clear measurement
instruments and study design reported
Full-text report was available in English.
Studies were excluded if:
1.
2.
3.
The articles were reviews, editorials or commentaries
They either comprised a single case study or had a
sample size \10
They were not deemed relevant to the primary focus on
the association between child heart disease and family
coping.
Data Extraction
Two authors independently evaluated and extracted the
data. The accuracy of extraction of each study was assessed
against the original document by a third author. The following data were extracted from included studies: (a) study
characteristics, including authors, date, the jurisdiction
where data were collected, characteristics of the children
and sample size; (b) method, including measurement
instruments, data sources and study design; (c) statistical
analysis employed; and (d) statistical data related to the
association between child heart disease and family coping.
Results
The review covers 25 studies with about half from European countries (N = 13) and the other half from the USA
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and Canada (N = 9) and one each from Egypt, Saudi
Arabia and Israel. All studies were conducted with families
of children with various forms of congenital heart disease
(CHD), ranging from mild to severe. Some studies included
comparison groups of families of children with other minor
illness and/or healthy children, whilst other studies compared families of children with mild CHD to those with
more complex or severe CHD. A PRISMA flow diagram
[44] shows the selection of papers for inclusion and
exclusion (Fig. 1) [42].
A majority of the studies employed quantitative measures (N = 17) with the use of control or comparison
groups. Four qualitative studies involved an exploratory
inquiry using semi-structured interviews and four utilised
both quantitative and qualitative methods. All quantitative
studies used at least one validated tool to measure various
factors pertaining to the familial impact of children with
CHD on parents. Details of studies examined are presented
in Table 1.
Discussion
Following a thematic analysis of the many factors that the
25 studies examined in relation to familial impact of child
heart disease and family coping, three major themes (along
with subthemes) were identified:
Fig. 1 PRISMA flow diagram
[44] of study selection process
I.
II.
III.
Impact on family: psychological distress and wellbeing; family functioning; quality of life
Coping: Psychological functioning; gender and age
differences
Parenting: parenting stress; parenting practices/style;
the role of attachment styles
Impact on Family
Psychological Distress and Well-being
The main findings on mental health outcomes for parents of
children with CHD (PCCHD) as compared to parents of
healthy children or children with other diseases generally
showed a higher incidence and severity of anger, anxiety,
distress, depression, hopelessness and/or somatisation symptoms [11, 38, 39]. The severity of the child’s heart lesion at
diagnosis was related to parental distress levels where
PCCHD with more severe lesions showed higher levels of
psychological distress [6]. Higher psychological distress was
also associated with poorer understanding of the diagnosis and
poorer cohesiveness in the family [13]. Gender differences
were observed for PCCHD, with mothers reporting higher
state anxiety [60], significantly more somatic symptoms [55]
and greater prevalence of clinically significant psychological
distress and hopelessness compared to fathers [13, 38].
Articles identified through multiple
databases search (n = 53, 506)
Articles screened by relevance
(n = 225)
Articles rejected at title/abstract or
duplicates (n = 82)
Additional records
identified (n = 42)
Full text articles retrieved for detailed
evaluation (n = 143)
Studies rejected (n = 160) with reasons:
1.
2.
3.
Studies meeting the inclusion criteria
4.
(n = 25)
5.
Review or editorials (n = 11)
Small sample size or single
case studies (n=5)
Sample does not include
families with CHD patients
under 14 (n =22)
Sample with other chronic
illness or conditions (n=25)
Studies with other focuses
such as impact of CHD on
child rather than family,
service provision,
measurement tools or program
evaluations (n =97)
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Study
Social impact on families
of children with complex
CHD
Quality of life among
PCCHD
Mothers’ attachment style,
their mental health, and
their children’s emotional
vulnerabilities: a 7-year
study of children with
CHD
References
Almesned et al. [1]
Arafa et al. [2]
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Berant et al. [3]
Children with heart diseases (67.5 %
CHD ? 32.5 % rheumatic heart
disease) of mean age 73.2 months
Children diagnosed with mild and
severe forms of CHD during their
first year of life
N = 63 Mothers/Israel
20 children with simple CHD and 21
children with complex CHD
managed in the univentricular tract
N = 41/Saudi Arabia
N = 400 ? 400 as comparison
group (parents of children with
minor illness)/Egypt
Child’s characteristics
Sample size/country
T3 (7 years post-diagnosis)—MHI
and marital satisfaction plus
children’s reported self-concept
and Children’s Apperception Test
T2 (1 year post-diagnosis)—MHI
and marital satisfaction (50-item
ENRICH)
T1 (diagnosis)—self-report
attachment style and Mental
Health Inventory (MHI)
A 7-year longitudinal study with
data collected on three occasions
with various measures:
Quantitative ? Qualitative
SF-36 V1 to assess health-related
quality of life (HRQL) on the
domains of physical functioning,
physical role, bodily pain,
general health, vitality, social
functioning, emotional role and
mental health
Structured questionnaire—sociodemographic characteristics,
CHD-related data, family-related
risk data;
Quantitative
Implications: Worthwhile considering the
role of parental attachment in shaping
children’s self-image and emotional wellbeing
Mothers’ attachment insecurities (both
anxiety and avoidance) at T1 were
associated with their children’s increased
emotional problems and poor selfimage 7 years later
Parametric tests, including correlations and
hierarchical regression analyses, revealed
that maternal avoidant attachment at T1
was the best predictor of deterioration in
the mothers’ mental health and marital
satisfaction over the 7-year period,
especially in a subgroup whose children
had severe CHD
Implications: Psychological status, social
support and reassurance of the parents to
address parental HRQOL should be
considered when making treatment
decision for their children
Factors found to have a significant impact
of HRQOL: severity of illness, type of
heart disease, age of child, having
multiple children, financial situation and
presence of comorbid condition. For
example, lowest mean scores for younger
age, rheumatic heart disease and female
children
MANOVA results revealed that parents of
children with heart diseases reported
significantly poorer HRQOL compared
with parents of children with minor
illness, except for the pain subscale, with
most significant differences on the
domains of general health, vitality and
physical role
Two-sample t test analyses revealed that
families of children with complex CHD
who underwent univentricular tract repair
had significantly higher IFS (61.3 vs.
52.0) mean scores than families with
simple CHD, particularly in the domain
of familial burden, followed by mastery,
financial burden and personal strain
Quantitative
Cross-sectional survey using the
24-item Impact on Family Scale
(IFS) administered to the two
groups of families. The IFS has
four subscales (on 4-point Likert
scale) assessing perceived 1.
financial burden; 2. familial/
social impact; 3. personal strain;
and 4. mastery
Key findings
Method/measures
Table 1 Details of studies that met the inclusion criteria for examining the relationship between congenital child heart disease and family impact and coping
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Study
Psychosocial outcomes for
preschool children and
families after surgery for
complex CHD
Psychological distress in
PCCHD: the impact of
prenatal versus post-natal
diagnosis
References
Brosig et al. [5]
Brosig et al. [6]
Child’s characteristics
Preschool-aged children (ages
3–6 years) with CHD, namely
hypoplastic left heart syndrome
(HLHS; N = 13) and transposition of
the great arteries (TGA; N = 13)
Ten infants prenatally diagnosed and
seven infants post-natally diagnosed
with severe CHD
Sample size/country
N = 31 parents/USA
N = 17 parents/USA
The Brief Symptom Inventory
(BSI)—completed at the time of
diagnosis, at the time of birth and
6 months after birth. Prenatal and
post-natal groups were compared
to each other and to BSI norms
Quantitative
Psychometric test scores were
compared to population norms
using z scores
Child Behaviour Checklist
Parent Behaviour Checklist
Parenting Stress Index
IFS
Paediatric quality of life inventory
Implication: need to provide parents with
psychological support, regardless of the
timing of diagnosis. Parents of children
with more severe lesions are at risk of
higher levels of psychological distress,
particularly over time
The severity of the child’s heart lesion at
diagnosis was related to parental distress
levels; parents with children with more
severe lesions had higher BSI scores (81
vs. 33 % with scores in the clinically
significant range)
Six months after birth, the post-natal group
scores on the BSI did not differ from test
norms, but the prenatal group scores
remained significantly higher than test
norms
Results using independent-sample t tests
revealed that both prenatal and post-natal
groups did not differ significantly from
each other on the BSI. Both groups’ BSI
was significantly higher than test norms
at the time of diagnosis
Implications: Importance of the
examination of parental stress, family
functioning, and behavioural
expectations for the child in the context
of routine medical/cardiac follow-up
Children with HLHS had higher rates of
attention (23 vs. 0 %) and externalising
behaviour problems (15 vs. 10 %) in the
clinical range than children with TGA
Parents of children with HLHS reported
greater parenting stress and more
negative impact of the child’s illness on
the family than parents of children with
TGA
PCCHD were more permissive in their
parenting style than parents of healthy
controls
Quality of life scores of PCCHD did not
differ from those of healthy controls
T tests and Mann–Whitney tests results
revealed:
Quantitative
Parents completed the following
measures:
Key findings
Method/measures
Table 1 Details of studies that met the inclusion criteria for examining the relationship between congenital child heart disease and family impact and coping
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123
Study
Mothers’ lived experiences
of support when living
with young children with
CHD
Maternal factors related to
parenting young children
with congenital heart
disease
The Meaning of Cost for
Families of Children
With CHD
References
Bruce et al.
[7]
Carey et al.
[8]
Connor
et al. [10]
Table 1 continued
Child’s characteristics
Children of 3–12 years old
diagnosed with CHD
Children (aged 2–5 years)
with moderate to severe
CHD
Children (aged 1 day to
5 years) with various
degrees of CHD complexity,
currently admitted for
congenital heart surgery
Sample size/country
N = 10 mothers/Sweden
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N = 30 ? 30 as control (all
mothers)/USA
N = 20 parents/USA
Exploratory inquiry using semi-structured interviews
(6 open-ended questions) focused on identifying
the meaning of ‘cost’ as experienced by the parents
and their perception of the social impact of these
costs on the family
Qualitative
Home visit by researchers to collect information on
family demographics, observation of parent–child
interaction and responses to a qualitative semistructured interview focusing on parenting
expectations, social support and help seeking
Quantitative ? Qualitative ? Observational?
Adopted a phenomenological-hermeneutic method
(naı̈ve understanding ? thematic structural
analysis ? comprehensive understanding) for
interpretation of the transcribed narrative
responses to a semi-structured interview format.
Interview length ranges from 50 min to 2 h long
Qualitative
Method/measures
Implication: Early intervention to brief
parents on additional stressors, e.g.
finances to be considered in overall
preparation
Complexity of the child’s disease and
parents’ SES was linked to higher levels
of stress experienced in the domains
identified
Thematic analysis revealed two major
categories of parents’ perceptions of the
cost burden associated with having a
child with CHD:(1) Lifestyle Changes
and (2) Uncertainty—across three
domains: financial, emotional and family
Implications: importance of promoting
normalisation among mothers of children
with CHD and ensuring a sufficient
support system
Qualitative data revealed mothers of CHD
children reporting high levels of
vigilance with their children
Quantitative (MANOVA) and parent–child
interaction results show similarity
between mothers of children with CHD
and controls in terms of parenting
practices and in their experiences of
parental stress related to childrearing
issues. Reported child behaviour was
similar across the two groups
Implications: mothers receiving personcentred and family-centred care feel more
supported and are more likely to adapt to
the stresses of parenting a child with
CHD
Mothers who feel supported were more
likely to adapt to the stresses of parenting
a child with CHD
Mothers receiving person-centred and
family-centred care reported feeling more
supported
Perception that improved coordination of
care and greater privacy would be
supportive
Need for practical support to overcome
limitations in daily life
Key themes identified in the qualitative
analysis included:
Key findings
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Child’s characteristics
Infants with mild (N = 92),
moderate (N = 50) or
severe CHD (N = 70)
Children with CHD (various
severity levels) aged
9 days–13.6 years
(M = 1.7 years)
Infants with significant CHD
Sample size/country
N = 212 mothers/Norway
N = 52 mothers/USA
N = 70 parents/UK
Study
Mothers of infants with
CHD: well-being from
pregnancy through the
child’s first 6 months
Psychological Adaptation
and Adjustment of
Mothers of Children
With CHD: Stress,
Coping, and Family
Functioning
Predictors of psychological
functioning in mothers
and fathers of infants
born with severe CHD
References
Dale et al.
[11]
Davis et al.
[12]
Doherty
et al. [13]
Table 1 continued
The 60-item situational version of COPE (The
Carver, Scheier and Weintraub’s multidimensional coping inventory)
The 53-item BSI (general severity index score);
Quantitative
Cognitive processes (maternal appraisal of stress,
expectations, methods of coping, family
functioning and maternal psychological adjustment
(Derogatis’ BSI))
Illness variables (15 prevalent types of heart defects)
Self- report inventories and a brief structured
interview that measure:
Quantitative ? Qualitative
Maternal mental health was predicted by
(1) maternal coping skills (:behavioural
disengagement : psychological distress);
(2) understanding of the diagnosis
(;understanding : psychological
distress); and (3) the degree of cohesive
family functioning (;cohesiveness :
psychological distress)
Mann–Whitney between-group
comparisons revealed elevated levels of
clinically significant psychological
distress in mothers, compared to fathers,
and differences between parents in
coping styles were found using
MANCOVA
Regression analyses showed after
controlling for maternal education and
defect severity, daily hassles and method
of coping accounted for a significant
amount of variance in maternal
adjustment scores. In contrast, family
supportiveness accounted for\1 % of the
remaining variance. All five variables
accounted for approximately 38 % of the
variance in maternal adjustment scores
Bivariate correlations reveal poorer
maternal adjustment (BSI scores) was
associated with high levels of daily stress
and palliative coping techniques and was
not significantly associated with severity
of the cardiac defect
Implication: The resilience of families is
underscored by this study. Need to
explore whether there is a risk of future
longer-term stressors in relation to the
child with CHD that will wear out
individual resources after the initial
period of mobilisation of forces to
achieve adaptation
Mothers of children with severe CHD
reported significantly more anger at
6 months post-partum
Parametric analyses revealed that mothers
of children with CHD showed a similar
level of satisfaction with life and feelings
of joy as mothers of children without
CHD both during pregnancy and when
the child was 6 months old
Quantitative
Longitudinal case-cohort design mothers completed
mailed questionnaires with assessment points at
gestation week 30 (T1) and at child’s age of
6 months (T2). Subjective well-being was
measured by The Satisfaction With Life Scale
(SWLS), The Joy and Anger scales (from
Differential Emotions Scale) and a single item on
social support
Key findings
Method/measures
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Study
Parent stress levels during
children’s hospital recovery after
congenital heart surgery
Quality of Life in Families of
Children with CHD
References
Franck et al. [14]
Goldbeck and
Melches [17]
Table 1 continued
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N = 69 patients and their
caregivers/Germany
N = 211 parents/UK
Sample size/country
Children aged 7–20 years of age
with CHD
Children 1 day–16 years of age
admitted for elective open and
closed heart surgery at a London
hospital
Child’s characteristics
Parents’ perceptions of their children’s
illness severity correlated with an
objective measure of post-operative
morbidity
Measures:
ULQIE for parental QL
LQ-KID-E for patient’s proxyreported QL completed by
parents
27-item LQ-KID for patients selfreported QL
Quality of Life (QL)—assessment:
Quantitative
Implication: Psychosocial interventions
should be family-focused and provide
support for patients’ and their caregivers’
QL
Significant interaction effect of parental QL
and patients’ self-reported QL in
predicting parental proxy reports on their
children’s QL. Parents with low own QL
agree significantly more with their
children than parents with high QL
Parental QL was correlated with both the
children’s self-rated QL and children’s
parent-rated QL
Children’s self-rated and proxy-rated QL
correlated moderately, with the highest
intra-class correlation on the subscale
psychological well-being/functioning
Paired Pearson correlations and regression
analyses revealed:
Implications: Better identification of
parents at risk of high stress and specific
interventions to improve parental support
and coping are needed pre- and postoperatively
Parents in more deprived communities and
mothers born outside the UK had higher
stress scores
Few differences between mothers’ and
fathers’ stress or their perceptions of their
children’s illness
Parental stress remained moderate to high
throughout their children’s
hospitalisation regardless of the severity
of illness
22-item self-report Parent Stressor
Scale: Infant Hospitalisation
(PSS-IH)
Parametric or nonparametric tests revealed
Parents completed questionnaires
and structured interviews
preoperatively and on postoperative days 3, 5, 8 and 15
Paternal mental health difficulties were
linked to the psychological factors of
worry and maladaptive coping strategies
such as disengagement through alcohol
use
Key findings
Quantitative
Method/measures
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CHD, parental stress and infant–
mother relationships
Predictors of Parental Quality of
Life after Child Open-Heart
Surgery: A 6-Month Prospective
Study
Goldberg
et al. [19]
Landolt
et al. [37]
Infants with CHD
Children (aged 0 to 16 years) with
CHD who had undergone openheart surgery
N = 42 ? 46 as
control (all mothers)/
Canada
N = 232 (135 mothers
and 97 fathers)/
Switzerland
Questionnaire-based data were collected at two
occasions: T1—discharge and T2—six
months after surgery. Measures included: SF36 to measure parent’s HRQoL; German
version of the 26-item Post-Traumatic
Distress Scale to assess surgery-related posttraumatic stress symptoms; 15-item Revised
Impact on Family Scale (IOS) to measure the
impact of the child’s disease on the family
Quantitative
Chi-square tests to compare data on measures
between CHD and healthy group. These
include: Infant–mother attachment
assessment using the strange situation to
classify dyads according to three major
patterns: secure dyad, insecure–avoidant
dyad and insecure–resistant dyad; Parenting
stress Index (120-item); Rutter Health
Questionnaire (24-item symptom checklist);
and child medical status
Quantitative ? Observational Assessment
Social risk factors—defined as single-parent
status, ethnic minority status, unfinished
parental education or professional training,
and/or unemployment
Ulm Inventory for Parents which measures
physical and daily functioning, satisfaction
with the situation in the family, emotional
stability, self-development and well-being
Symptoms of PTSD at discharge and a high
IOS score at 6 months were associated with
lower mental component scores on HRQoL
in both parents
All impaired domains of parental HRQoL at
discharge significantly improved over time
and were within or above population norms
6 months later
Mothers had lower HRQoL than fathers
Both mothers and father showed lower levels of
HRQoL at discharge in domains such as role
limitations due to physical and emotional
problems
A range of statistical analyses were performed
including Chi-square analyses for nominal
variables comparisons; one-sample Student’s
t tests for testing between-group differences;
and Wilcoxon matched-pair signed-rank tests
to compare scores at T1 vs. T2
Securely attached infants showed more
subsequent improvement in health than
insecurely attached infants showed
No significant differences between the secure
and insecure infants with CHD on parental
stress or well-being measure
Chi-square tests indicated that infants with
CHD were less likely to be securely attached
to their mothers than the healthy group. More
avoidant babies were identified in the CHD
group
Implication: Programmes providing
psychosocial support for children with CHD
and their caregivers should consider risk
factors which are both medical and social
A significant interaction effect indicated a
cumulative negative impact of disease
severity and social disadvantage on the
quality of life of the patients
Significant effects of social disadvantage on the
quality of life of both the children and their
parents
Significant effects of disease severity on
children’s’ quality of life
Three measures were employed:
*Severity of the disease was
evaluated by the responsible
paediatrician
ANOVA results demonstrated:
Quantitative
Children with various severities* of
CHD with ages ranged from 0 to
21 years
N = 132 families of
children with CHD/
Germany
The impact of the severity of
disease and social disadvantage
on quality of life in families with
CHD.
Goldbeck
and
Melches
[18]
Ulm Inventory for Children which measures
physical well-being, psychological wellbeing, disease- and therapy-related distress,
family relations and general quality of life
perception
Key findings
Method/measures
Child’s characteristics
Sample size/country
Study
References
Table 1 continued
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123
Study
Distress and hopelessness
among PCCHD and parents of
children with other diseases,
and parents of healthy
children
Psychosocial morbidity among
PCCHD: A prospective
longitudinal study
References
Lawoko and
Soares [38]
Lawoko and
Soares [39]
Table 1 continued
123
Quantitative
Children born with CHD between 0
and 20 years of age who were
living at home, with parents with
responsible for their care or in
close contact with them during
the survey
Children between 0 and 20 years of
age with CHD
N = 1092 PCCHD ? 112 parents
of children with other diseases
(PCOD) ? 293 randomised
healthy control group (PHC)/
Sweden
N = 632 parents/Sweden
Longitudinal quantitative design,
data collected twice, a year apart.
Repeated measures covering (1)
children’s demographic and
clinical characteristics; (2)
Caregiving time/burden (use of
Symptom Check List- Revised
(SCL-90-R) and Hopelessness
Scale to assess psychosocial
morbidity); (3) 12-item Schedule
for Social interaction; (4)15-item
Pyramid Patient Questionnaire
and 8-item Client Satisfaction
Questionnaire; (5) Parental
financial situation
Cross-sectional questionnaire
administration in a 20-day period
with measures on distress (SCL90-R) and hopelessness (20-item
Hopelessness Scale)
Method/measures
Child’s characteristics
Sample size/country
Long-standing psychosocial morbidity (at both
T1 and T2) was also relatively high
(7–22 %)
Univariate analyses including independentsample t test, paired-sample t test, and Chisquare test plus blockwise logistic regression
analyses were performed. High point
prevalence of depression, anxiety,
somatisation, and hopelessness symptoms
among PCCHD both at T1 (14–31 %) and T2
(15–38 %); Higher severity of symptoms
reported by mothers than fathers
Implications: Programme interventions should
be targeted at parents, in particular PCCHD
who are at risk of developing psychosocial
problems
Variables such as employment status and
financial situation explained more of the
variation in distress and hopelessness among
parents than the diseases of their children
There were no differences in distress and
hopelessness between PCOD and PHC
Fathers of children with CHD had higher
distress than fathers belonging to the other
groups
Mothers within all parent groups had higher
levels of distress and hopelessness than
fathers, with the highest levels among
mothers of children with CHD compared to
mothers in the other groups
PCCHD had higher scores on distress and
hopelessness than PCOD and PHC. A
significant number of parents, in particular
PCCHD, reported levels of distress and
hopelessness within/above psychiatric
outpatients and depressed people,
respectively
Parametric and nonparametric tests (ANOVAs,
Chi-square tests, t tests, Dunn’s/Bonferroni
post hoc tests and blockwise logistic
regression analyses) results revealed:
Implications: Parents in whom the child’s CHD
has a high impact on the family are at
increased risk of persistent low mental
HRQoL
Key findings
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N = 53 (25 parents ? 28
grandparents)/Canada
Facets of Parenting a Child with
HLHS.
Patients with univentricular heart in
early childhood: parenting stress
and child behaviour
Rempel
et al.
[45]
Sarajuuri
et al.
[49]
N = 37 parents of
newborns with
CHD ? 46 parents of
healthy control
(gender-matched
newborns)/Finland
Children age 6 months–4.5 years
who had undergone surgery for
CHD (N = 15)
N = 53 (25 parents ? 28
grandparents)/Canada
Parenting under Pressure: a
grounded theory of parenting
young children with lifethreatening CHD
Rempel
et al.
[46]
23 newborns with HLHS and 14
newborns with univentricular
heart defects (UVH)
Children age 6 months–4.5 years
who had undergone surgery for
HLHS. (N = 15)
Child’s characteristics
Sample size/country
Study
References
Table 1 continued
Parenting Stress Index (PSI)
Child Behaviour Checklist (CBCL)
Measures:
Quantitative
A review of descriptive and intervention studies
in CHD
Knowledge from clinical practice in nursing
children with HLHS
Conceptual memos written by the researchers
as part of the analytic process,
Transcriptions ? audio recordings, and field
notes of the 53 previously conducted
interviews,
Four data sources were used to conceptualise
the parenting needs for young children with
CHD:
Qualitative and Observational (Secondary
analysis of data from Study 19)
Telephone and in-person recorded interviews
were conducted. Participants were asked to
recount experiences from the time of the
child’s diagnosis to the present
Using a constructive grounded theory study
design, data collection and data analysis
occurred simultaneously (using NVivo 8) to
capture a full range of parenting experiences
from multiple family members and from each
stage of the surgical trajectory.
Qualitative
Method/measures
Parents of children with HLHS reported
significantly more total and internalising
behaviour problems than the controls. For the
syndrome scales, a significant difference was
only found in somatic complaints
Statistical analyses using independent- and
paired-sample t tests revealed significantly
higher PSI mean score among the mothers
and fathers of children with HLHS compared
to controls
Supported parenting (We did what we could do
to help)
Uncertain parenting (We didn’t know what to
expect)
Expert parenting (She became a nurse)
‘Hands-off’ parenting (There’s nothing we
could do)
Survival parenting (It will test you to the
utmost)
Interpretation of the data led to the
conceptualisation of a model of five facets of
parenting of children with CHD. Each facet
constitutes a critical component for
educational or psychosocial intervention for
parents:
Implications: Interventions that focus on
helping carers to move through the four
phases may help them safeguard the survival
of their children, and their own survival as
parents as they manage multiple demands
(4) encountering new challenges
(3) watching for and accommodating the
unexpected; and
(2) becoming increasingly attached;
(1) realising and adjusting to the inconceivable;
Qualitative analysis revealed four overlapping
and re-emerging phases:
Implication: Psychosocial intervention may be
beneficial
Higher psychosocial morbidity associated with
parental caregiving burden, dissatisfaction
with care, social isolation, and financial
instability rather than children’s clinical
severity
Key findings
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Children under age 12 with newly
diagnosed CHD (within the last
3–4 months of the study)
N = 92 two-parent
families/USA
Family stress, perceived social
support and coping following the
diagnosis of a child’s CHD
Tak and
McCubbin
[57]
The Coping Health Inventory for Parents
(CHIP)
Personal Resources Questionnaire (PRQ-85)
Implications: Interventions developed for
PCCHD need to focus on building
resilience through social support
Child and family characteristics (such as
child’s age and family stress) were
significant predictors of perceived social
support and parental coping
Perceived social support operated as a
resiliency factor between family stress
and both parental and family coping
The Family Inventory of Life Events (FILE)
Regression Analyses revealed:
Both parents independently completed
instruments on:
Mothers also appeared to seek social
support more often compared to fathers
PCCHD showed weaker tendency to use
styles of coping such as reassuring
thoughts and less often expressed
negative emotions (anger, annoyance)
compared to the reference group as
measured by UCL
Statistical analyses performed using onesample t tests and univariate analyses of
(co)variance. Results showed parents of
children treated for CHD had lower levels
of distress, manifested as lower levels of
somatic symptoms, anxiety and
sleeplessness and serious depression
compared with reference group.
Favourable long-term outcomes may be
due to the good to excellent prognosis of
the CHD conditions that this sample had.
The authors did highlight that overall this
sample did not require further reinterventions or reoperations and hence
the results may not be considered
representative for parents of patients with
complex malformations. Mothers of
children with CHD reported significantly
more somatic symptoms than fathers
Implications: The stress reported by parents
indicates the need for early psychosocial
support for the families of these seriously
ill patients
The parents of patients with UVH did not
differ from the controls in terms of PSI
score or reported CBCL scores
Key findings
Quantitative
The Utrecht Coping List (UCL) (7-scales)
The 28-item version of General Health
Questionnaire (assess levels of psychological
distress);
Quantitative
Children and adolescents aged from
7 to 15 years who underwent
invasive treatment for CHD
N = 109 (52 sets of both
parents; 48 mothers and
9 fathers)/Netherlands
Long-term psychological distress,
and styles of coping, in parents of
children and adolescents who
underwent invasive treatment for
CHD
Spijkerboer
et al. [55]
Method/measures
Child’s characteristics
Sample size/country
Study
References
Table 1 continued
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Child’s characteristics
Children who underwent openheart surgery over the period
2002 through 2007 with mean
age at assessment of
45.3 months.
Children aged from birth to
16.9 years
Sample size/country
N = 114 mothers ? 82
fathers/Netherlands
N = 3 groups of parents of
75 children who—(1.
undergone surgery for
CHD (PCCHD); 2.
transplantation of bone
marrow (PCOD); 3.
healthy (PHC))/UK
Study
A multi-centric study of diseaserelated stress, and perceived
vulnerability, in parents of
children with CHD
Psychological functioning in
parents of children undergoing
elective cardiac surgery
References
VrijmoetWiersma
et al. [60]
Wray and
Sensky
[62]
Table 1 continued
Utrecht coping list
Dyadic adjustment scale
General health questionnaire
Measures:
Parents of the children undergoing cardiac
surgery or transplantation of bone marrow
were assessed before the procedure and again
12 months after these procedures, whilst the
healthy group were assessed on two
occasions, 12 months apart.
Quantitative
Child Vulnerability Scale.
State-Trait Anxiety Index
Parental Stress Index-Short Form
General Health Questionnaire
The Paediatric Inventory for Parents (short
form)
Few differences between any of the groups
on the other parameters, and the
evaluated indexes showed stability over
time
Following treatment, there was a significant
reduction in the levels of distress in
PCCHD and PCOD
PCCHD and PCOD both had significantly
higher rates of distress prior to the
surgical procedures than did PHC
Results using combination of parametric
and nonparametric tests revealed:
Implications: Psychosocial screening of
parents of children with CHD is
important in order to provide appropriate
counselling to those parents most in need
Severity of the lesion did not influence
parental levels of stress, but parents of
children with HLHS reported higher
levels of stress than other parents
Risk factors for increased anxiety and
perceived vulnerability included number
of surgical procedures, time since the last
procedure, and ethnicity
PCCHD reported significantly higher rates
of perceived vulnerability than did
parents of healthy children
State anxiety levels were higher in mothers
of children with CHD.
Parents of children with CHD did not report
higher generic nor disease-related stress
scores, and parenting levels of stress were
also comparable to reference groups
Statistical analysis using independent
t tests, ANOVA and forced entered
regression analyses were performed to
compare parental responses with the
reference groups on the instruments used
and to determine the predictors of
perceived vulnerability and parenting
stress. Analyses showed:
Quantitative
Cross-sectional study using both generic and
disease-related measures for assessment:
Key findings
Method/measures
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The timing of diagnosis and treatment also seemed to play
a role in the findings reported. For example, Wray and
Sensky [62] found that following CHD treatment, there was
a significant reduction in the levels of distress in PCCHD.
Moreover, prenatal versus post-natal diagnosis of CHD
seems to impact on the psychological distress in PCCHD.
Contrary to their hypothesis, Brosig et al. [6] found that the
level of distress for parents in the prenatal group remained
significantly higher than test norms from diagnosis to
6 months after birth. Considering the financial burden
placed on families of children with CHD, Connor et al. [10]
in their qualitative study noted that prenatal diagnosis could
trigger financial uncertainty in family members and often
resulted in altered personal spending prior to birth.
In terms of mental health outcomes for PCCHD, Dale
et al., [11] reported a somewhat different pattern of findings. Using measures of subjective well-being rather than
clinical psychological assessment tools, the study found
similar levels of life satisfaction and positive emotions
such as feelings of joy in mothers of children with and
without CHD, both during pregnancy and at 6 months postpartum.
Family Functioning
Complexity of the child’s disease and parents’ socioeconomic status were linked to higher levels of family uncertainty and more drastic changes in lifestyles where the
impact on family functioning went beyond monetary issues.
Families of children with more severe CHD (e.g. hypoplastic left heart syndrome—HLHS) reported more negative
impact than families of children with milder forms of CHD,
particularly in the domains of familial burden (including
more problems with siblings), social relationships, mastery,
financial burden and personal strain [1, 5, 10].
Nevertheless, parental caregiving burden, dissatisfaction
with care, social isolation and financial instability may be
more important for PCCHD’s psychosocial experiences than
the sole presence and severity of CHD in children [39]. For
example, according to some studies, socioeconomic stressors such as unemployment and financial strain explained
more of the variation in psychosocial experiences among
PCCHD than the diseases of their children [14, 18, 38].
Quality of Life
Research on the quality of life (QoL) of PCCHD (i.e. individual’s subjective perception of physical, psychological
and social functioning) produced contrasting findings where
different approaches and measurement tools were used with
different samples. For example, similar QoL scores were
found between parents of different CHD diagnostic groups
(HLHS vs. TGA) and healthy controls using the Paediatric
123
Quality of Life Inventory [5]. Another study, however, using
Medical Outcomes Study Short Form 36 (SF-36) revealed
that PCCHD (particularly for parents with younger age,
rheumatic heart disease and female children) reported significantly poorer health-related quality of life (HRQoL). The
most significant differences were found in the domains of
general health, vitality and physical role limitation [2].
Some QoL studies have utilised multi-informant strategies including proxy and self-reports to integrate different
viewpoints which allows a more comprehensive assessment of QoL of the whole family over time. Landolt et al.
[37] found that mothers showed lower HRQoL than fathers
at discharge. At 6 months after surgery, a higher impact of
the child’s disease on family life was a risk factor for a low
mental HRQoL in both mothers and fathers. Parents’ selfreported QoL is also shown to be associated with children’s
self-reported HRQoL [17].
Coping
Psychological Functioning
In the literature reviewed, several studies used the transactional stress and coping model to better understand the
psychological functioning of families and coping at different stages of the diagnosis/treatment and in both mothers
and fathers. A lower degree of maternal health and
adjustment was found to be associated with maladaptive
coping strategies (e.g. avoidance/behavioural disengagement, denial/wishful thinking) but was unrelated to the
severity of heart defects [12, 13]. Perceived social support
was found to be an important factor that positively related
to individual parental coping [57].
Gender and Age Differences
Gender and age differences were found between styles of
coping in PCCHD. Mothers appeared to ‘vent’ more about
the situation, seek more social support (both instrumental
and emotional) and avail themselves of more spiritual and
religious support compared to fathers, who used alcohol
significantly more than mothers [13, 55]. Younger mothers
reported more helpful coping related to family integration,
cooperation and optimism, whereas for younger fathers
helpful coping was related to maintaining social support,
self-esteem and psychological stability [57].
Parenting
Parenting Stress
One study employing the Parent Stressor Scale: Infant
Hospitalisation found that the stress levels of PCCHD
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remained moderate to high throughout their children’s
hospitalisation regardless of the severity of illness [14].
The general consensus of the parental stress studies was
that parent stress levels related to child rearing more generally, as distinct from the specific period of hospitalisation, did not differ between PCCHD and healthy controls.
However, within the CHD group, parents of children with
HLHS reported more parenting stress than parents of
children with minor forms of heart defect (e.g. TGA) [33,
36, 50, 53]. Parenting children with HLHS who are at risk
of mortality and morbidity explained the high levels of
pressure and stress that this group of parents experienced as
they have to constantly watch for and accommodate the
uncertainties and challenges that arise in their parenting
journey [45].
Parenting Practices/Style
Contrasting findings in parenting styles using the Parent
Behaviour Checklist (PBC) were reported in the studies
reviewed. Some reported more permissive styles of parenting in PCCHD than in the parents of healthy controls
[5], whereas others showed similarity in nurturing and
discipline practices by mothers in both the CHD and
healthy control groups. The contrasting findings are consistent with Rempel et al. [45] 5-facet parenting practices
model which articulates the diverse and multi-faceted
experiences and needs of PCCHD.
The Role of Attachment Style
The studies also identified the protective role of attachment
security for both the parent raising a child with CHD and
the child themselves. More instances of avoidant attachment style were identified in the CHD group as compared
to the healthy control group. Those infants who were
securely attached to their mothers showed more subsequent
improvement in health than insecurely attached infants
[19]. Avoidant mothers of CHD children, particularly those
with severe levels of CHD, were found to experience the
greatest deterioration in mental health and marital satisfaction over time with impacts on their children’s emotional issues and self-image [3]. These studies highlight the
importance of supporting mothers living with young children with CHD by providing person-centred and familycentred care, reinforcing the findings of Bruce et al. [7].
Conclusion and Implications for Clinical Practice
This review of the familial impact of CHD and parenting
practices of PCCHD did not yield conclusive results.
However, there was a consensus that PCCHD who have
fewer psychosocial resources and less support may be at
risk of higher psychological distress and lower well-being,
for both the parent and the child and that this impact may
persist over time. This review generally supports the
assertion by Jackson et al. [26] that the psychosocial
impacts on families of children with complex conditions
may be determined by individual and family factors
beyond the severity of the child’s condition. Parenting
coping programmes for children with HLHS were indicated, however. The findings on familial coping were
consistent with previous findings on the importance of
helping families to identify existing coping patterns and
parenting practices particularly at the diagnostic stage [24,
28]. Familial factors such as cohesiveness and adaptive
parental coping strategies appear to be paramount for
successful parental adaptation to CHD in their child, which
in turn plays a significant role in parents’ and children’s
long-term well-being. The resilience of families of children
with CHD is underscored, although normalising parental
anxiety and developing interventions to manage this anxiety is highlighted, along with the need to monitor familial
impacts and coping over time to identify those parents who
may need continuing or renewed support.
Assessment of familial impact may be achieved through
the use of a psychosocial assessment measure, in recognition
of the multiplicity of factors determining impact and coping.
A useful example of such a measure is the Psychosocial
Assessment Tool (PAT2.0), developed for use with families
of paediatric cancer patients [43]. This tool, which appears
to be modifiable for CHD families, assesses psychosocial
risk within a number of domains: family structure and
resources, family social support, family problems, parent
stress reactions, family beliefs, child problems and sibling
problems. Such a measure could be administered at prenatal
or post-natal diagnosis, at first or subsequent major surgeries, and post-psychosocial intervention.
The review implies a holistic approach in the paediatric
care of CHD children and their families through understanding and addressing the diverse and multi-faceted
experiences and needs of PCCHD. Early psychosocial
intervention programmes for PCCHD should go beyond the
examination of parental stress and family functioning to
include specific interventions targeting improved adaptive
coping and enhanced productive parenting practices. These
interventions should also be aimed at reducing structural or
systemic contributors to impaired coping and poorer
attachment outcomes. Interventions to promote coping
should target promotion of maternal attachment in the
immediate post-diagnosis period, particularly where children have severe CHD.
Monitoring of familial impact and parental coping over
time, through the use of a CHD version of the PAT 2.0, or
similar tool is warranted. Added to this is monitoring of
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how parents cope in the hospital environment, a factor
known to be important in familial adjustment to serious
childhood medical conditions [27]. The four-phase model
[46] which suggests that parents need to realise and adjust
to the inconceivable; become increasingly attached; watch
for and accommodate the unexpected; and encounter and
deal with new challenges provides a useful guide for health
professionals working with PCCHD in gauging the
capacity of parents to adjust and cope.
The use of a validated psychosocial risk screen for all
PCCHD would enable the early identification of those
parents and families who are at risk, and their allocation to
the most relevant psychosocial intervention protocol, based
on a framework such as the pediatric psychosocial preventative health model (PPPHM) [29, 33]. This model
builds on the assumption that most families can cope with
their child’s condition, but that some will need assistance
to deal with their distress and to possibly learn new coping
and parenting skills. Using public health concepts, the
PPPHM, based on a validated risk assessment, will allocate
families who are transiently distressed but basically resilient, to universal services, such as psycho-education;
families with acute distress and other risk factors, such as
avoidant forms of coping, to targeted services; and families
with multiple original or escalating risk factors, to clinical
services [9]. This screening and differential diagnosis of
level of psychosocial risk can streamline allocation of
services and enhance the specificity of service provision in
health services with limited resources.
As with acute care, the severity of psychosocial risk
demands a proportional response in terms of number and
composition of a multidisciplinary team to provide the
appropriate intervention [53]. In paediatric cancer settings,
universal care typically involves the medical team with
access to allied health, for example, on request [41]. In a
PPPHM-oriented service, targeted and clinical interventions may involve a paediatric nephrologist, nurse clinician, dietician, social worker and psychologist [41]. A
paediatric cardiac multidisciplinary team, in addition to the
cardiologist, may include the same range of allied health
professionals, with adjustments according to the level of
risk. In some cases, universal interventions may require a
health psychologist, targeted interventions may require a
neuropsychologist, whilst some clinical interventions may
require a clinical psychologist or family therapist, especially if dealing with pre-existing comorbidities in parents
and family members or dealing with acute distress with
dysfunctional familial coping patterns or dysfunctional
parenting style.
The approach suggested here of universal screening and
selective multidisciplinary intervention requires a strong
organisational commitment, rather than just an individual
clinician or departmental commitment. It can be justified,
123
however, in terms of its contribution to secondary prevention and increased family and child well-being.
Acknowledgments This review was undertaken as part of a larger
study, the Heart Child Family Project, conducted by the Heart
Research Centre (HRC) and the Melbourne Graduate School of
Education, University of Melbourne, funded by HeartKids Australia
under the 2014 Grants-in-Aid program.
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