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.
Highlights
It is well documented that mothers of children with intellectual disabilities or autism experience elevated stress, with mental health compromised
Participants For all of the syndrome groups included in the study with the exception of Rett syndrome (RTT) (AS, Cornelia de Lange syndrome (CdLS), Down syndrome (DS), Fragile-X syndrome (FXS), Phelan-McDermid [Phelan McDermid syndrome (PMS)], Prader-Willi [Prader-Willi syndrome (PWS)], Rubinstein-Taybi [Rubenstein Taybi syndrome (RTS)], Soto, Smith-Magenis [Smith Magenis syndrome (SMS)], Tuberous Sclerosis Complex [TSC], 1p36 deletion syndrome, 8p23 deletion syndrome and the autism group [Autism Spectrum Disorder (ASD)]), questionnaire packs were distributed to members of each syndrome support group within the UK. [27] provides detailed information on the recruitment process but briefly, questionnaire packs were provided to the syndrome support groups who sent them out to their members
The results of the Bayesian model averaging (BMA) indicate two groups based upon syndrome, with Group 1 reporting significantly lower positive gain than Group 2
Summary
It is well documented that mothers of children with intellectual disabilities or autism experience elevated stress, with mental health compromised. Mothers of children with neurodevelopmental disorders and/or intellectual and developmental disabilities (IDD) report elevated levels of stress and affective symptoms when compared to mothers of typically developing children [1,2,3]. These differences in maternal well-being are established by the time children are 3–5 years of age [3, 4] and continue over time [5]. The limited studies that have included or focused upon fathers draw mixed conclusions with regards to levels of stress compared to mothers and compared to fathers of typically developing children [8]
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