دورية أكاديمية

Sex bias in autism spectrum disorder in neurofibromatosis type 1

التفاصيل البيبلوغرافية
العنوان: Sex bias in autism spectrum disorder in neurofibromatosis type 1
المؤلفون: Garg, S, Heuvelman, H, Huson, S, Tobin, H, Green, J, Network, Northern UK NF1 Research
بيانات النشر: BioMed Central
سنة النشر: 2018
المجموعة: Oxford University Research Archive (ORA)
الوصف: Background Despite extensive literature, little is known about the mechanisms underlying sex bias in autism spectrum disorder (ASD). This study investigates the sex differences in ASD associated with neurofibromatosis type 1, a single-gene model of syndromic autism. Methods We analysed data from n = 194 children aged 4–16 years with neurofibromatosis type 1. Sex differences were evaluated across the Autism Diagnostic Interview-Revised (ADI-R), Autism Diagnostic Observation Schedule (ADOS), verbal IQ, Social Responsiveness Scale (SRS) and Conners questionnaires. Results There was 2.68:1 male:female ratio in children meeting ASD criteria on the deep phenotyping measures. On symptom profile, males with neurofibromatosis type 1 (NF1) + ASD were more impaired on reciprocal social interaction and communication domains of the ADI-R but we found no differences on the restricted, repetitive behaviours (RRBs) domain of the ADI-R and no differences on the social on the ADOS. NF1 ASD males and females were comparable on verbal IQ, and the inattention/hyperactivity domains of the Conners questionnaire. Conclusions There is a significant male bias in the prevalence of ASD in NF1. The phenotypic profile of NF1 + ASD cases includes greater social communication impairment in males. We discuss the implications of our findings and the rationale for using NF1 as a model for investigating sex bias in idiopathic ASD.
نوع الوثيقة: article in journal/newspaper
اللغة: English
العلاقة: https://ora.ox.ac.uk/objects/uuid:28974f48-8631-424f-a0b1-12b7ac141678Test; https://doi.org/10.1186/s11689-016-9159-4Test
DOI: 10.1186/s11689-016-9159-4
الإتاحة: https://doi.org/10.1186/s11689-016-9159-4Test
https://ora.ox.ac.uk/objects/uuid:28974f48-8631-424f-a0b1-12b7ac141678Test
حقوق: info:eu-repo/semantics/openAccess ; CC Attribution (CC BY)
رقم الانضمام: edsbas.D1A60095
قاعدة البيانات: BASE