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dc.contributor.authorAdams, Dawn
dc.contributor.authorHastings, Richard P
dc.contributor.authorAlston-Knox, Clair
dc.contributor.authorCianfaglione, Rina
dc.contributor.authorEden, Kate
dc.contributor.authorFelce, David
dc.contributor.authorGriffith, Gemma
dc.contributor.authorMoss, Jo
dc.contributor.authorStinton, Chris
dc.contributor.authorOliver, Chris
dc.date.accessioned2019-06-19T13:01:43Z
dc.date.available2019-06-19T13:01:43Z
dc.date.issued2018
dc.identifier.issn1750-1172
dc.identifier.doi10.1186/s13023-018-0924-1
dc.identifier.urihttp://hdl.handle.net/10072/381061
dc.description.abstractBackground: It 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. Methods: Mothers 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. Results: Different 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. Conclusions: Bayesian 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.
dc.description.peerreviewedYes
dc.languageEnglish
dc.publisherBioMed Central
dc.publisher.placeUnited Kingdom
dc.relation.ispartofchapter185
dc.relation.ispartofpagefrom1
dc.relation.ispartofpageto14
dc.relation.ispartofjournalOrphanet Journal of Rare Diseases
dc.relation.ispartofvolume13
dc.subject.fieldofresearchSpecial Education and Disability
dc.subject.fieldofresearchEducational Psychology
dc.subject.fieldofresearchOther Medical and Health Sciences
dc.subject.fieldofresearchcode130312
dc.subject.fieldofresearchcode170103
dc.subject.fieldofresearchcode1199
dc.titleUsing 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
dc.typeJournal article
dc.type.descriptionC1 - Articles
dc.type.codeC - Journal Articles
dc.description.versionPublished
gro.facultyArts, Education & Law Group, School of Education and Professional Studies
gro.hasfulltextFull Text
gro.griffith.authorAlston-Knox, Clair L.
gro.griffith.authorAdams, Dawn M.


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