ArticleOrphanet journal of rare diseases2018
Using Bayesian methodology to explore the profile of mental health and well-being in 646 mothers of children with 13 rare genetic syndromes in relation to mothers of children with autism.
Article in Orphanet journal of rare diseases, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers, 2 of them syntheses that pooled it.
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Who cites it
10 citing papers in PubMed, 2 syntheses or guidelines pooled it.
- The burden of illness in Prader-Willi syndrome: a systematic literature review.Orphanet journal of rare diseases · 2025Pooled it
- Factors and experiences that influence school mobility for autistic students: A systematic review.The British journal of educational psychology · 2022Pooled it
- Association between a diagnostic journey in Angelman syndrome and caregivers' quality of life.Journal of neurodevelopmental disorders · 2026Article
- Measuring economic burden in families of individuals with Angelman Syndrome in Poland: a caregivers' survey.Orphanet journal of rare diseases · 2025Article
- Exploring the psychological problems of mothers having the experience of death of children with untreatable genetic disorders: A qualitative study.Journal of education and health promotion · 2024Article
- A systematic review of in-patient psychiatric care for people with intellectual disabilities and/or autism: effectiveness, patient safety and experience.BJPsych open · 2022Review
- Psychological distress and positive gain in mothers of children with autism, with or without other children with neurodevelopmental disorders.International journal of developmental disabilities · 2022Article
- Failure to replicate a robust Down syndrome advantage for maternal well-being.Journal of intellectual disability research : JIDR · 2021Article
- The Relationship Between Child Anxiety and the Quality of Life of Children, and Parents of Children, on the Autism Spectrum.Journal of autism and developmental disorders · 2020Article
- The Developmental Behaviour Checklist (DBC) Profile in Young Children on the Autism Spectrum: The Impact of Child and Family Factors.Journal of autism and developmental disorders · 2019Article
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10 authors.
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Abstract
backgroundIt is well documented that mothers of children with intellectual disabilities or autism experience elevated stress, with mental health compromised. However, comparatively little is known about mothers of children with rare genetic syndromes. This study describes mental health and well-being in mothers of children with 13 rare genetic syndromes and contrasts the results with mothers of children with autism.
methodsMothers of children with 13 genetic syndromes (n = 646; Angelman, Cornelia de Lange, Down, Fragile-X, Phelan McDermid, Prader-Willi, Rett, Rubenstein Taybi, Smith Magenis, Soto, Tuberous Sclerosis Complex, 1p36 deletion and 8p23 deletion syndromes) and mothers of children with autism (n = 66) completed measures of positive mental health, stress and depression. Using Bayesian methodology, the influence of syndrome, child ability, and mother and child age were explored in relation to each outcome. Bayesian Model Averaging was used to explore maternal depression, positive gain and positive affect, and maternal stress was tested using an ordinal probit regression model.
resultsDifferent child and mother factors influenced different aspects of mental well-being, and critically, the importance of these factors differed between syndromes. Maternal depression was influenced by child ability in only four syndromes, with the other syndromes reporting elevated or lower levels of maternal depression regardless of child factors. Maternal stress showed a more complex pattern of interaction with child ability, and for some groups, child age. Within positive mental health, mother and child age were more influential than child ability. Some syndromes reported comparable levels of depression (SMS, 1p36, CdLS) and stress (SMS, AS) to mothers of children with autism.
conclusionsBayesian methodology was used in a novel manner to explore factors that explain variability in mental health amongst mothers of children with rare genetic disorders. Significant proportions of mothers of children with specific genetic syndromes experienced levels of depression and stress similar to those reported by mothers of children with autism. Identifying such high-risk mothers allows for potential early intervention and the implementation of support structures.
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