Chi, Z;
Devine, RT;
Wolstencroft, J;
Skuse, D;
Hughes, C;
Baker, K;
(2024)
Rare neurodevelopmental conditions and parents’ mental health – how and when does genetic diagnosis matter?
Orphanet Journal of Rare Diseases
, 19
(1)
, Article 70. 10.1186/s13023-024-03076-2.
Preview |
PDF
s13023-024-03076-2.pdf - Published Version Download (2MB) | Preview |
Abstract
Background: Parents of individuals with rare neurodevelopmental conditions and intellectual disabilities (ID) are vulnerable to mental health difficulties, which vary between parents and within parents over time. The underlying cause of a child’s condition can influence parents’ mental health, via uncertain pathways and within unknown time-windows. Results: We analysed baseline data from the IMAGINE-ID cohort, comprising 2655 parents of children and young people with ID of known genetic origin. First, we conducted a factor analysis of the SDQ Impact scale to isolate specific pathways from genetic aetiology to parents’ mental health. This suggested a two-factor structure for the SDQ Impact scale, with a “home & distress” dimension and a “participation” dimension. Second, we tested via structural equation modelling (SEM) whether genetic diagnosis affects Impact and mental health directly, or indirectly via children’s characteristics. This analysis identified an indirect pathway linking genetic aetiology to parents’ mental health, serially through child characteristics (physical disabilities, emotional and behavioural difficulties) and Impact: home & distress. Third, we conducted moderation analysis to explore the influence of time elapsed since genetic diagnosis. This showed that the serial mediation model was moderated by time since diagnosis, with strongest mediating effects among recently diagnosed cases. Conclusions: There are multiple steps on the pathway from ID-associated genetic diagnoses to parents’ mental health. Pathway links are strongest within 5 years of receiving a genetic diagnosis, highlighting opportunities for better post-diagnostic support. Recognition and enhanced support for children’s physical and behavioural needs might reduce impact on family life, ameliorating parents’ vulnerabilities to mental health difficulties.
| Type: | Article |
|---|---|
| Title: | Rare neurodevelopmental conditions and parents’ mental health – how and when does genetic diagnosis matter? |
| Location: | England |
| Open access status: | An open access version is available from UCL Discovery |
| DOI: | 10.1186/s13023-024-03076-2 |
| Publisher version: | http://dx.doi.org/10.1186/s13023-024-03076-2 |
| Language: | English |
| Additional information: | This work is licensed under a Creative Commons Attribution 4.0 International License. The images or other third-party material in this article are included in the Creative Commons license, unless indicated otherwise in the credit line; if the material is not included under the Creative Commons license, users will need to obtain permission from the license holder to reproduce the material. To view a copy of this license, visit http://creativecommons.org/licenses/by/4.0/ |
| Keywords: | Carers, Genetic, Intellectual disability, Mental health, Parents |
| UCL classification: | UCL UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Population Health Sciences > UCL GOS Institute of Child Health > Population, Policy and Practice Dept |
| URI: | https://discovery-pp.ucl.ac.uk/id/eprint/10188262 |
Archive Staff Only
 |
View Item |
