Christina Manning, Peter L Hurd, Silven Read, Bernard Crespi
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Males, but not females, showed significant genotypic effects for the SPQ-BR subscale associated with speech and language: Odd Speech. These findings, in conjunction with animal model studies showing vocalization and auditory effects of SHANK3 mutations, and studies indicating severe language alterations and speech-associated white matter tract abnormalities in Phelan-McDermid syndrome, suggest that SHANK3 differentially affects the development and expression of human language and speech. Imaging genetic and speech-language studies of typical individuals carrying different genotypes of rs9616915 should provide novel insights into the neurological and psychological bases of speech and language alterations among individuals with SHANK3 mutations and Phelan-McDermid syndrome.</p>","PeriodicalId":8659,"journal":{"name":"Autism Research and Treatment","volume":"2021 ","pages":"6634584"},"PeriodicalIF":0.0000,"publicationDate":"2021-06-03","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8195663/pdf/","citationCount":"0","resultStr":"{\"title\":\"SHANK3 Genotype Mediates Speech and Language Phenotypes in a Nonclinical Population.\",\"authors\":\"Christina Manning, Peter L Hurd, Silven Read, Bernard Crespi\",\"doi\":\"10.1155/2021/6634584\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Mutations affecting the synaptic-scaffold gene SHANK3 represent the most common genetic causes of autism with intellectual disability, accounting for about 1-2% of cases. Rare variants of this gene have also been associated with schizophrenia, and its deletion results in the autistic condition known as Phelan-McDermid syndrome. Despite the importance of SHANK3 as a paradigmatic gene mediating neurodevelopmental disorders, its psychological effects in nonclinical populations have yet to be studied. We genotyped the nonsynonymous, functional SHANK3 SNP rs9616915 in a large population of typical individuals scored for autism spectrum traits (the Autism Quotient, AQ) and schizotypy spectrum traits (the Schizotypal Personality Questionnaire, SPQ-BR). Males, but not females, showed significant genotypic effects for the SPQ-BR subscale associated with speech and language: Odd Speech. These findings, in conjunction with animal model studies showing vocalization and auditory effects of SHANK3 mutations, and studies indicating severe language alterations and speech-associated white matter tract abnormalities in Phelan-McDermid syndrome, suggest that SHANK3 differentially affects the development and expression of human language and speech. Imaging genetic and speech-language studies of typical individuals carrying different genotypes of rs9616915 should provide novel insights into the neurological and psychological bases of speech and language alterations among individuals with SHANK3 mutations and Phelan-McDermid syndrome.</p>\",\"PeriodicalId\":8659,\"journal\":{\"name\":\"Autism Research and Treatment\",\"volume\":\"2021 \",\"pages\":\"6634584\"},\"PeriodicalIF\":0.0000,\"publicationDate\":\"2021-06-03\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8195663/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Autism Research and Treatment\",\"FirstCategoryId\":\"1085\",\"ListUrlMain\":\"https://doi.org/10.1155/2021/6634584\",\"RegionNum\":0,\"RegionCategory\":null,\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"2021/1/1 0:00:00\",\"PubModel\":\"eCollection\",\"JCR\":\"\",\"JCRName\":\"\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Autism Research and Treatment","FirstCategoryId":"1085","ListUrlMain":"https://doi.org/10.1155/2021/6634584","RegionNum":0,"RegionCategory":null,"ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2021/1/1 0:00:00","PubModel":"eCollection","JCR":"","JCRName":"","Score":null,"Total":0}
引用次数: 0
摘要
影响突触支架基因SHANK3的突变是自闭症伴智力残疾最常见的遗传原因,约占病例的1-2%。这种基因的罕见变异也与精神分裂症有关,它的缺失会导致被称为费伦-麦克德米德综合征的自闭症。尽管SHANK3作为介导神经发育障碍的典型基因具有重要意义,但其在非临床人群中的心理影响尚未得到研究。我们在大量自闭症谱系特征(自闭症商数,AQ)和分裂型人格问卷(SPQ-BR)得分的典型个体中对非同音的功能性SHANK3 SNP rs9616915进行了基因分型。在言语和语言相关的SPQ-BR子量表中,男性表现出显著的基因型效应,而女性则没有。这些发现,结合动物模型研究表明SHANK3突变对发声和听觉的影响,以及研究表明Phelan-McDermid综合征中严重的语言改变和语言相关的白质道异常,表明SHANK3对人类语言和言语的发育和表达有不同的影响。对携带不同rs9616915基因型的典型个体进行影像学遗传和言语语言研究,将为SHANK3突变和Phelan-McDermid综合征患者言语和语言改变的神经学和心理学基础提供新的见解。
SHANK3 Genotype Mediates Speech and Language Phenotypes in a Nonclinical Population.
Mutations affecting the synaptic-scaffold gene SHANK3 represent the most common genetic causes of autism with intellectual disability, accounting for about 1-2% of cases. Rare variants of this gene have also been associated with schizophrenia, and its deletion results in the autistic condition known as Phelan-McDermid syndrome. Despite the importance of SHANK3 as a paradigmatic gene mediating neurodevelopmental disorders, its psychological effects in nonclinical populations have yet to be studied. We genotyped the nonsynonymous, functional SHANK3 SNP rs9616915 in a large population of typical individuals scored for autism spectrum traits (the Autism Quotient, AQ) and schizotypy spectrum traits (the Schizotypal Personality Questionnaire, SPQ-BR). Males, but not females, showed significant genotypic effects for the SPQ-BR subscale associated with speech and language: Odd Speech. These findings, in conjunction with animal model studies showing vocalization and auditory effects of SHANK3 mutations, and studies indicating severe language alterations and speech-associated white matter tract abnormalities in Phelan-McDermid syndrome, suggest that SHANK3 differentially affects the development and expression of human language and speech. Imaging genetic and speech-language studies of typical individuals carrying different genotypes of rs9616915 should provide novel insights into the neurological and psychological bases of speech and language alterations among individuals with SHANK3 mutations and Phelan-McDermid syndrome.