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dc.contributor.authorGeoffray, Marie-Maude
dc.contributor.authorRobinson, Louise
dc.contributor.authorRamamurthy, Kavitha
dc.contributor.authorManderson, Lauren
dc.contributor.authorO'Flaherty, Julieta
dc.contributor.authorLehtonen, Annukka
dc.contributor.authorTordjman, Sylvie
dc.contributor.authorGreen, Jonathan
dc.contributor.authorVassallo, Grace
dc.contributor.authorGarg, Shruti; email: shruti.garg@manchester.ac.uk
dc.date.accessioned2021-07-12T01:08:41Z
dc.date.available2021-07-12T01:08:41Z
dc.date.issued2021-06-12
dc.date.submitted2021-02-22
dc.identifierpubmed: 34182240
dc.identifierpii: S0022-3956(21)00376-9
dc.identifierdoi: 10.1016/j.jpsychires.2021.06.002
dc.identifier.citationJournal of psychiatric research, volume 140, page 545-550
dc.identifier.urihttp://hdl.handle.net/10034/625224
dc.descriptionFrom PubMed via Jisc Publications Router
dc.descriptionHistory: received 2021-02-22, revised 2021-05-17, accepted 2021-06-04
dc.descriptionPublication status: aheadofprint
dc.description.abstractThe impact of the Neurofibromatosis type 1 (NF1) on cognition have been subject to much clinical investigation, but environmental modifiers of disease expression have not yet been systematically investigated. The aim of this paper is to determine the role of demographic and environmental factors such as age, sex, socioeconomic status, parental NF1 status and neurological complications on the cognitive, behavioural and academic outcomes in NF1. Participants included 206 children aged 4-18 years seen within the Manchester clinical research NF1 service. Multiple linear regression models were used to study the effect of the hypothesized predictor variables on cognitive, behavioural and academic outcomes. Relative to population norms, 80% of the NF1 sample demonstrated significantly lower scores in at least one cognitive, behavioural or academic domains. Family history of NF1 and lower SES were independently associated with poorer cognitive, behavioural and academic outcomes. Neurological problems such as epilepsy and hydrocephalus were associated with lower IQ and academic skills. Cognitive and behavioural phenotypes emerge commonly via a complex interplay between genes and environmental factors, and this is true also of a monogenic condition such as NF1. Early interventions and remedial education may be targeted to risk groups such those with familial NF1, families with lower SES and those with associated neurological comorbidities. [Abstract copyright: Copyright © 2021 Elsevier Ltd. All rights reserved.]
dc.languageeng
dc.sourceeissn: 1879-1379
dc.subjectCognitive and behavioural development
dc.subjectNF1
dc.subjectSocio-economic status
dc.titlePredictors of cognitive, behavioural and academic difficulties in NF1.
dc.typearticle
dc.date.updated2021-07-12T01:08:41Z
dc.date.accepted2021-06-04


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