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dc.contributor.authorWolfe, K
dc.contributor.authorStrydom, A
dc.contributor.authorMorrogh, D
dc.contributor.authorCarter, J
dc.contributor.authorCutajar, P
dc.contributor.authorEyeoyibo, M
dc.contributor.authorHassiotis, A
dc.contributor.authorMcCarthy, J
dc.contributor.authorMukherjee, R
dc.contributor.authorPaschos, D
dc.contributor.authorPerumal, N
dc.contributor.authorRead, S
dc.contributor.authorShankar, Rohit
dc.contributor.authorSharif, S
dc.contributor.authorThirulokachandran, S
dc.contributor.authorThygesen, JH
dc.contributor.authorPatch, C
dc.contributor.authorOgilvie, C
dc.contributor.authorFlinter, F
dc.contributor.authorBardi, Ugo
dc.contributor.authorBass, N
dc.date.accessioned2021-05-23T10:51:42Z
dc.date.available2021-05-23T10:51:42Z
dc.date.issued2017-01
dc.identifier.issn1018-4813
dc.identifier.issn1476-5438
dc.identifier.urihttp://hdl.handle.net/10026.1/17196
dc.description.abstract

Chromosomal copy-number variations (CNVs) are a class of genetic variants highly implicated in the aetiology of neurodevelopmental disorders, including intellectual disabilities (ID), schizophrenia and autism spectrum disorders (ASD). Yet the majority of adults with idiopathic ID presenting to psychiatric services have not been tested for CNVs. We undertook genome-wide chromosomal microarray analysis (CMA) of 202 adults with idiopathic ID recruited from community and in-patient ID psychiatry services across England. CNV pathogenicity was assessed using standard clinical diagnostic methods and participants underwent comprehensive medical and psychiatric phenotyping. We found an 11% yield of likely pathogenic CNVs (22/202). CNVs at recurrent loci, including the 15q11-q13 and 16p11.2-p13.11 regions were most frequently observed. We observed an increased frequency of 16p11.2 duplications compared with those reported in single-disorder cohorts. CNVs were also identified in genes known to effect neurodevelopment, namely NRXN1 and GRIN2B. Furthermore deletions at 2q13, 12q21.2-21.31 and 19q13.32, and duplications at 4p16.3, 13q32.3-33.3 and Xq24-25 were observed. Routine CMA in ID psychiatry could uncover ~11% new genetic diagnoses with potential implications for patient management. We advocate greater consideration of CMA in the assessment of adults with idiopathic ID presenting to psychiatry services.

dc.format.extent66-72
dc.format.mediumPrint-Electronic
dc.languageen
dc.language.isoeng
dc.publisherSpringer Science and Business Media LLC
dc.rightsAttribution-NonCommercial-NoDerivatives 4.0 International
dc.rightsAttribution-NonCommercial-NoDerivatives 4.0 International
dc.rightsAttribution-NonCommercial-NoDerivatives 4.0 International
dc.rightsAttribution-NonCommercial-NoDerivatives 4.0 International
dc.rightsAttribution-NonCommercial-NoDerivatives 4.0 International
dc.rights.urihttp://creativecommons.org/licenses/by-nc-nd/4.0/
dc.rights.urihttp://creativecommons.org/licenses/by-nc-nd/4.0/
dc.rights.urihttp://creativecommons.org/licenses/by-nc-nd/4.0/
dc.rights.urihttp://creativecommons.org/licenses/by-nc-nd/4.0/
dc.rights.urihttp://creativecommons.org/licenses/by-nc-nd/4.0/
dc.subjectAdolescent
dc.subjectAdult
dc.subjectAged
dc.subjectAutism Spectrum Disorder
dc.subjectCalcium-Binding Proteins
dc.subjectCell Adhesion Molecules, Neuronal
dc.subjectChromosome Aberrations
dc.subjectChromosomes, Human, Pair 15
dc.subjectChromosomes, Human, Pair 16
dc.subjectComparative Genomic Hybridization
dc.subjectDNA Copy Number Variations
dc.subjectEngland
dc.subjectFemale
dc.subjectHumans
dc.subjectIntellectual Disability
dc.subjectMale
dc.subjectMicroarray Analysis
dc.subjectMiddle Aged
dc.subjectNerve Tissue Proteins
dc.subjectNeural Cell Adhesion Molecules
dc.subjectReceptors, N-Methyl-D-Aspartate
dc.subjectSchizophrenia
dc.subjectWhite People
dc.titleChromosomal microarray testing in adults with intellectual disability presenting with comorbid psychiatric disorders
dc.typejournal-article
dc.typeJournal Article
plymouth.author-urlhttps://www.webofscience.com/api/gateway?GWVersion=2&SrcApp=PARTNER_APP&SrcAuth=LinksAMR&KeyUT=WOS:000394116100012&DestLinkType=FullRecord&DestApp=ALL_WOS&UsrCustomerID=11bb513d99f797142bcfeffcc58ea008
plymouth.issue1
plymouth.volume25
plymouth.publication-statusPublished
plymouth.journalEuropean Journal of Human Genetics
dc.identifier.doi10.1038/ejhg.2016.107
plymouth.organisational-group/Plymouth
plymouth.organisational-group/Plymouth/Faculty of Health
plymouth.organisational-group/Plymouth/Users by role
dc.publisher.placeEngland
dcterms.dateAccepted2016-06-28
dc.identifier.eissn1476-5438
dc.rights.embargoperiodNot known
rioxxterms.versionofrecord10.1038/ejhg.2016.107
rioxxterms.licenseref.urihttp://creativecommons.org/licenses/by-nc-nd/4.0/
rioxxterms.licenseref.startdate2016-01
rioxxterms.typeJournal Article/Review


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