Social functioning and mental health in children with CHD
Research type
Research Study
Full title
Clinically significant correlates of social functioning and mental health in children with congenital heart disease: pilot study
IRAS ID
310010
Contact name
Vicky Elliott
Contact email
Sponsor organisation
Leicestershire Partnership NHS Trust
Duration of Study in the UK
0 years, 5 months, 27 days
Research summary
Research Summary
Background
Many children born with complex congenital heart disease (CHD) live active, happy lives following surgery, but concerns remain regarding long-term outcomes. Some have conditions, (e.g. 22q11.2 deletion syndrome, 22q11DS), associated with learning and mental health difficulties. However, children with CHD unaccompanied by genetic abnormalities may also experience such difficulties. Further, outcomes vary substantially within both groups. This pilot study will investigate the feasibility of a larger study of factors contributing to variability in outcomes. Identifying these will assist services to identify “at risk” children earlier and provide preventive interventions.
Aims
The proposed larger study aims to:
1. Compare social development and mental health in four groups:
-Children with “simple” transposition of the great arteries, a CHD rarely associated with a genetic syndrome
-Children with CHD and 22q11DS, a genetic condition conferring increased risk for developmental and mental health difficulties
-Children with 22q11DS but without CHD
-Children with neither CHD nor 22q11DS
2. Examine how treatments received, child temperament, and family environment interact with CHD and 22q11DS to influence social development and mental health.
The aim of the pilot is to evaluate the feasibility of the larger study with respect to participant recruitment and acceptability of study procedures.
Methods
Five families in each group, with children aged 3-15, will be invited to participate.
Using parental interviews, questionnaires, and review of medical records, we will collect information on children’s:
-Treatment history (e.g. scheduling of operations)
-Development, using structured scales
-Mental health, using parent-completed screening assessments
-Temperament, i.e. early personality
and demands on parents and parenting practices.
Participants will be recruited via clinical staff at the East Midlands Congenital Heart Centre and Leicestershire Clinical Genetics Service. Participation will involve parents completing questionnaires (estimated time required 2 hours) and one interview and one summary/debrief meeting with a researcher (estimated total time 90 minutes).Summary of Results
This research was carried out by a clinical psychologist (Dr. Vicky Elliott) and a consultant paediatric cardiologist (Professor Frances Bu’Lock), who both work at the East Midlands Congenital Heart Centre (EMCHC), with support from an honorary researcher from Leicester University (Dr. Fred Furniss). The study was sponsored by the Leicestershire Partnership NHS Trust, and carried out with the co-operation of the University Hospitals of Leicester NHS Trust. The research received no financial support from any other source.
The research was planned and carried out with the support of eleven Parent Advisers from Heart Link, the East Midlands charity for parents and families with children with congenital heart disease (CHD). The Parent Advisers, all of whom have a child or grandchild with CHD, had a major role in the design of the research, especially regarding factors which may influence mental health outcomes for children with CHD. They also advised on data collection and methods for feeding back to participants.
The study took place between November 2023 and December 2024 at the EMCHC in Leicester.
We thank the participating families, the Parent Advisers, and the Trustees and Committee members of Heart Link for their contributions to, and support for, this project.
Purpose of the study.
Developments in surgical techniques over the last 30-40 years have improved outcomes and survival for children with complex CHD. For some of these children, their CHD is caused by the presence of a genetic syndrome which itself causes difficulties with development and mental health. However, some children who have CHD without an associated genetic syndrome still experience difficulties with various aspects of cognitive functioning and mental health. The long-term purpose of this research is to look for ways of identifying, at an early age, those children who are most at risk of developing difficulties later on, so that services can provide earlier support.
This study was a pilot study to identify any potential difficulties before carrying out a larger study. It concentrated on two groups of young people, those with CHD which is rarely seen associated with a genetic condition (“simple” transposition of the great arteries, or TGA) and those with CHD associated with 22q11.2 deletion syndrome (22q11.2DS) a genetic condition linked to increased risk for developmental and mental health difficulties. It also included two comparison groups of children who do not have CHD;
· children with 22q11DS but no CHD,
· children with neither CHD nor any identified genetic syndrome.
The primary aim of the pilot study was to answer the following questions
· would sufficient families be interested in participating in such research
· would parents be willing to complete the required number of questionnaires concerning their child’s development and mental health
Study method
Who participated in the study?
Clinical staff at the East Midlands Congenital Heart Centre and the Leicester Clinical Genetics Services were asked to identify potential participants and to supply the family with a copy of the Project Information Sheet and ask whether their contact details could be passed to the research team.
A member of the research team contacted interested parents to explain more about the study, and arranged to meet parents who consented to be involved. A total of seven children (two with transposition of the great arteries, two with 22q11.2 deletion syndrome, and three with no genetic condition or CHD) participated in the research.
What happened during the study?
Participating parents met with a researcher, either face-to-face or via video link, to complete a questionnaire to assess various aspects of their child’s development. Parents were asked about aspects of their child's treatment history and were asked for written permission for a researcher to review relevant medical records. The researcher also asked about the child’s friendships, and then completed with the parent(s) two brief questionnaires, one covering their experiences of the challenges of caring for a child with a medical condition, and one about their “parenting style”. Parents were then invited to return and complete further questionnaires (with the support of the researcher if requested). These included questionnaires about:
- the child’s overall mental health, possible symptoms of post-traumatic stress, and anxiety
- the child’s activity levels and ability to maintain concentration
- the child’s social development including symptoms of possible autism spectrum conditions
- extent and type of any bullying the child had experienced
- the child’s early personality, including their sociability, sensitivity, and ability to control their emotions.
What were the results of the study?
Recruitment of families
Overall, we succeeded in recruiting less than 50% of our target number of participants.
Three families out of a total of 22 families including a child with 22q11.2 DS who were contacted by letter from the Leicester clinical genetics service agreed to participate in the research, a positive response rate of 14%: however, one later withdrew.
Two families with children with TGA agreed to participate in the research. We do not know how many families were informed about the research during face-to-face contacts with clinicians, but we do know that only one family agreed to participate following such an approach. When EMCHC clinical staff wrote to families of children with TGA, one out of 10 families approached agreed to participate in the research, a response rate of 10%.
Two of the five families who participated in the study agreed also to complete questionnaires on a total of three of their other children who had neither CHD nor 22q11.2DS,
Completion of questionnaires
All but one of the participating parents completed and returned usable questionnaire packs (as defined in the protocol) within two weeks.
How has this study helped patients and researchers?
The study was intended to be a pilot project to explore problems that might be encountered in carrying out a larger project looking for ways to identify those children with CHD who are most at risk of developing mental health difficulties. The pilot study has provided valuable information for planning of future research.
For a larger study to be meaningful, we would need to recruit a minimum of 13 families in each of the four groups. Given the response rate from families of children with 22q11.2DS in the pilot study, this would suggest that we would need to approach approximately 180-280 families with children with 22q11.2 DS in order to find sufficient families interested in participating. Similarly, the results of the pilot study suggest that we would need to contact 130 families of children with TGA in order to carry out a larger study.
We had also been concerned that potential participants might be put off participating in the research by the prospect of completing the large number of questionnaires involved, particularly the seven questionnaires which participants completed on their own. In fact however all the families who completed the questionnaires found them straightforward and completed them all in well under an hour.
How have the results of the study been shared with participants and stakeholders?
Each family who participated in the study was provided with a summary of the results of the study (in which only group-level data was included), and participating parents were invited to an online meeting to discuss the results with the researchers. The summary was also sent to the Parent Advisers and the Heart Link committee.
Details of any further research planned
We are currently reviewing plans for further research in the light of the findings of the pilot study, particularly with regard to the recruitment of participants. We had already expected that in order to recruit sufficient families of children with 22q11.2DS for a larger study, we would need the cooperation of other clinical genetics services outside of Leicestershire. We had however expected that we would be able to recruit sufficient families of children with TGA through the EMCHC. Our experience with the pilot project suggests that this may not be possible, and that to carry out a larger project may require cooperation with other specialist children’s cardiac services.
Where can I learn more about this study? For further information about this study, please contact Dr. Vicky Elliott (vicky.elliott1@nhs.net) or Dr. Fred Furniss (fgf1@leicester.ac.uk).
REC name
North West - Greater Manchester East Research Ethics Committee
REC reference
23/NW/0156
Date of REC Opinion
2 Aug 2023
REC opinion
Further Information Favourable Opinion