The CHIP-Family intervention for young children with congenital heart disease and their family: a randomized controlled trial

(Please also see METC protocol par. 1, which also includes references)

Accumulating evidence demonstrates that children with congenital heart disease
(CHD) are at increased risk for behavioral, emotional, and cognitive problems
in childhood and adulthood. In line with international research, our previous
cohort studies have indicated that CHD-children, compared with healthy
children, are two times more likely to develop psychopathology (16-27% vs.
10%); this was irrespective of the type of the cardiac defect. Especially
internalizing problems (anxiety, depression), problems with social contacts,
and reduced quality of life have been reported, which may hamper school
functioning. Moreover, neuropsychological problems are well known in these
children. Our previous studies have demonstrated intellectual impairments and
elevated percentages of patients that attended special education (24% vs. 4 %
in norm). This has long-term consequences; a long-term cohort study of our
research group has shown that adults with CHD overall had a lower occupational
and educational status and lower income compared with the general population.
The combination of behavioral/emotional problems, developmental delay, and
school difficulties, represents the most common morbidity affecting the quality
of life in school-aged survivors of CHD [9]. Furthermore, children with CHD
often have reduced stamina and participate less in exercise and sports. This
has a negative impact on their quality of life.
Research has shown that parental (especially maternal) factors play a crucial
role in children*s psychosocial wellbeing. Maternal mental health and worry
have appeared to be more important predictors of children*s psychosocial
wellbeing than illness severity. Unfortunately, parents of CHD-children are
also at risk for psychosocial problems (1 year prevalence 7-22%; e.g. anxiety,
depression). Most studies have documented on psychosocial morbidity in mothers,
while fathers have been largely neglected. Clinical experience learns that
fathers play a crucial role in family functioning. Since parental psychosocial
functioning is an important mediator in children*s wellbeing, a family-based
psychosocial intervention is very important to prevent or minimize mental
health problems of CHD-children and their parents, and also to strengthen
children*s emotional resilience.
For parents and children with CHD, key milestones in life present more barriers
and challenges than for families with healthy children. Such key developmental
milestones are: starting kindergarten at 4 and 5 years of age (a major step
towards *letting go* for parents and towards autonomy for children) and
starting primary school at 6 and 7 years of age (structurally places cognitive
demands and pressures on the child). Considering the emotional and cognitive
vulnerability of CHD-children in these milestone periods, their reduced
exercise capacity, and the need expressed by 40% of parents in our institution
for a psychosocial intervention, it is very important to develop a
family-centered intervention in which the child and both parents play a central
role. Through such an intervention, psychological problems of children with CHD
and their parents can be reduced or prevented, and school functioning,
emotional resilience, and sport participation of children can be improved. This
can improve the quality of life of children and also that of their parents,
enabling them to coach their children more adequately. It can also enhance
mental and physical health of children with CHD and their siblings, and the
participation in social activities. Therefore, this study focuses on further
developing and testing such a psychosocial intervention of 4 to 7 year old
children with CHD and their families.
Until now, the only published evidence-based intervention tailored to the
developmental transition of starting school for young children with CHD, is the
Congenital Heart Disease Intervention Program * School (CHIP-School). The
CHIP-School study focused on promoting psychosocial wellbeing of preschoolers
with CHD and their mothers. Recently, the efficacy of CHIP-School was proven,
with significant gains in: maternal mental health, reduced perceived strain on
the family, and less days *sick*/less school absence of the child. However, as
to child psychosocial wellbeing, only a non-significant, though positive, trend
was found.
Shortcomings of CHIP-School were that neither a separate child module to
enhance child emotional resilience, nor fathers were included. In collaboration
with the CHIP-developer (prof. McCusker, Belfast) we will innovate, strengthen
and extend CHIP-School, thus developing a new program: CHIP-Family.
In the new program, CHIP-Family, 1) a tailored child module to further improve
children*s wellbeing and 2) fathers (multi-informant approach) will be
included. Also, 3) the social validity of CHIP-Family (i.e. how parents
evaluate the intervention; satisfaction) and its separate elements will be
studied in order to identify the most beneficial ingredients.

The objective of this study is to investigate:
1) the effectiveness of CHIP-Family on psychosocial wellbeing (i.e.: behavioral
and emotional problems) in 4 to 7 year old children who underwent cardiac
surgery or a catheter intrervention for CHD.
2) the effectiveness of CHIP-Family on parental mental health (for both mothers
and fathers) of parents of these children.
3) the effectiveness of CHIP-Family on:
- psychosocial wellbeing of the CHD-children, consisting of: school
functioning, sports participation, and quality of life;
- quality of life of their brothers/sisters.
4) which psychosocial and medical factors can predict the success of
CHIP-Family.

(Please also see METC protocol par. 3 and fig. 1)

This is a single-center, single blinded, randomized controlled trial (RCT). The
baseline assessment (T1) will take place within 2 months after starting
kindergarten or primary school. Families will then be randomly allocated -
stratified for age and CHD severity - to the CHIP-Family or the care as usual
(CAU) group. All patients will receive adequate medical care. Patients who are
randomized to the CHIP-Family group will also complete a social validity
assessment within 2 weeks after the intervention. The post-assessment (T2) will
take place 6 months after T1.

It was explicitly chosen to do the follow-up assessment 6 months after T1
(instead of direct post-test assessment immediately after CHIP-Family) because:
1) we want to test the effect of CHIP-Family on psychosocial well-being and
school functioning over a longer period of time (this has more clinical
relevance in our opinion than a direct post-assessment).
2) we want to avoid a *test effect*; if children and parents remember the items
of the questionnaires after such a short interval, this will increase the
likelihood of a test effect.
3) several questionnaires explicitly ask in their instructions how a child or
adult has felt over a longer period of time. Thus, repeating the questionnaires
after a too short interval (a direct post-assessment) is not possible without
compromising the psychometric basis, and therefore contra-indicated.

(Please also see METC protocol par. 5)

The CHIP-Family intervention consists of:
- A one-day (6 hour) group workshop for mothers and fathers (problem
prevention, psycho-education, parenting skills), plus for each parent couple an
*individual* follow-up session (1 hours, ± 4 weeks later). In each group, 7 to
11 parents take part.
- A separate one-day (6 hours) children's workshop to promote emotional
resilience and fun in sports. In each group 7 to 11 children plus one sibling
per child take part. The children*s workshop takes place simultaneously with
the parent workshop. To keep the size of the children*s group workshop
realistically feasible to work with and to have a comparable amount of
participating siblings per child, only one sibling per child can participate in
this group workshop.

The new children's module (group workshops and individual follow-up sessions)
includes:
- Child-friendly, playful exercises, based on the evidence-based FUN
FRIENDS!-protocol. This FUN FRIENDS!-protocol is the only cognitive behavioral
therapy protocol for 4- to 7-year-olds in the Netherlands and has been
published by the dept. of Child and Adolescent Psychiatry/Psychology of the
Erasmus MC.The cognitive behavioral therapy-exercises focus specifically on
themes related to CHD, such as: sports and exercise tolerance, relaxation,
promoting autonomy, strengthening self-esteem, making friends, problem solving
skills and positive thinking.
- Sports exercises, taught by an experienced physiotherapist. The exercises are
based on a standardized training program. Previous research of our team has
shown that these exercises are effective in improving the quality of life of
children with CHD.

CHIP-Family will be performed in a standardized manner by a clinical
psychologist (parent workshops/sessions), a pediatric cardiologist (parent
workshop), a junior psychologist (child workshops/sessions), and an experienced
physiotherapist (child workshops). The junior psychologist will be assisted by
master*s students in Psychology (interns). A one-day CHIP-Family training will
be given to the psychologists by expert prof. McCusker, developmental
psychologist, Queens University Belfast, developer and investigator of the
CHIP-protocol.

Care as usual: After randomization, half of the patients will receive the
CHIP-Family intervention; the other half will receive their regular medical
care, called care as usual (thus no additional psychosocial intervention).

The risks associated with participation can be considered negligible and the
burden can be considered minimal.

At present, there is no regular psychosocial intervention available for young
children with CHD and their families; the majority of families receive no
psychosocial care at all. Children with CHD are at increased risk for
behavioral, emotional, and cognitive problems in childhood and adulthood. Their
parents are also at risk for psychosocial problems. Therefore, psychotherapy
will benefit them. Previous research has shown beneficial effects of the
CHIP-School intervention. The module added to CHIP-Family are based on the
evidence-based FUN FRIENDS!-protocol. Moreover, the added sports exercises have
been proven to improve quality of life. Therefore, we hypothesize that the
CHIP-Family will result in better child psychosocial wellbeing (i.e. less
emotional/behavioral problems) and improved parental mental health.
If patients and their parents do not receive CHIP-Family, but remain in the CAU
group, no harm is done (i.e. compared to the routine treatment which is
currently provided). If parents/patients state that they are in need for acute
psychosocial care, or if the research psychologist identifies an acute need for
psychosocial care, adequate referral will be arranged.

Parents and teachers will be individually asked to report on psychosocial
topics via web based questionnaires. This will take approximately 2 hours for
parents per assessment, 5 minutes for children and 30 minutes for teachers.
Families who are randomly allocated to the CHIP-Family condition, will attend a
6 hour workshop and a parents will attend a 1 hour follow-up session.