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dc.contributor.authorAdams, Dawnen_US
dc.contributor.authorHastings, Richarden_US
dc.contributor.authorAlston-Knox, Clairen_US
dc.contributor.authorCianfaglione, Rinaen_US
dc.contributor.authorEden, Kateen_US
dc.contributor.authorFelce, Daviden_US
dc.contributor.authorGriffith, Gemmaen_US
dc.contributor.authorMoss, Joen_US
dc.contributor.authorStinton, Chrisen_US
dc.contributor.authorOliver, Chrisen_US
dc.date.accessioned2019-06-19T13:01:43Z
dc.date.available2019-06-19T13:01:43Z
dc.date.issued2018en_US
dc.identifier.issn1750-1172en_US
dc.identifier.doi10.1186/s13023-018-0924-1en_US
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.en_US
dc.description.peerreviewedYesen_US
dc.languageEnglishen_US
dc.publisherBioMed Centralen_US
dc.publisher.placeUnited Kingdomen_US
dc.relation.ispartofchapter185en_US
dc.relation.ispartofpagefrom1en_US
dc.relation.ispartofpageto14en_US
dc.relation.ispartofjournalOrphanet Journal of Rare Diseasesen_US
dc.relation.ispartofvolume13en_US
dc.subject.fieldofresearchSpecial Education and Disabilityen_US
dc.subject.fieldofresearchEducational Psychologyen_US
dc.subject.fieldofresearchOther Medical and Health Sciencesen_US
dc.subject.fieldofresearchcode130312en_US
dc.subject.fieldofresearchcode170103en_US
dc.subject.fieldofresearchcode1199en_US
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 autismen_US
dc.typeJournal articleen_US
dc.type.descriptionC1 - Articlesen_US
dc.type.codeC - Journal Articlesen_US
dc.description.versionPublisheden_US
gro.facultyArts, Education & Law Group, School of Education and Professional Studiesen_US
gro.hasfulltextFull Text


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