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雷特综合征女童的言语语言能力方面受MECP2突变类型的影响。

Aspects of speech-language abilities are influenced by MECP2 mutation type in girls with Rett syndrome.

作者信息

Urbanowicz Anna, Downs Jenny, Girdler Sonya, Ciccone Natalie, Leonard Helen

机构信息

Telethon Kids Institute, The University of Western Australia, Australia; School of Exercise and Health Sciences, Edith Cowan University, Perth, WA, Australia.

出版信息

Am J Med Genet A. 2015 Feb;167A(2):354-62. doi: 10.1002/ajmg.a.36871. Epub 2014 Nov 26.

Abstract

This study investigates relationships between methyl-CpG-binding protein 2 gene (MECP2) mutation type and speech-language abilities in girls with Rett syndrome. Cross-sectional data on 766 girls, aged 15 years and under, with genetically confirmed Rett syndrome was obtained from the Australian Rett Syndrome Database (ARSD) (n = 244) and the International Rett Syndrome Phenotype Database (InterRett) (n = 522). Relationships between MECP2 mutation type and age of regression in speech-language abilities, and the level of speech-language abilities before and after this regression were investigated. The females had a median age of 4.95 years in the ARSD and 5.25 years in InterRett. The majority (89%, 685/766) acquired speech-language abilities in the form of babble or words at some point in time. Of those who acquired babble or words, 85% (581/685) experienced a regression in these abilities. Those with a p.Arg133Cys mutation were the most likely to use one or more words, prior to (RRR = 3.45; 95% CI 1.15-10.41) and after (RRR = 5.99; 95% CI 2.00-17.92), speech-language regression. Girls with Rett syndrome vary in their use of speech and language, and in their experience of speech-language regression and these variations are partly explained by genotype.

摘要

本研究调查了患有雷特综合征女孩的甲基化CpG结合蛋白2基因(MECP2)突变类型与言语语言能力之间的关系。从澳大利亚雷特综合征数据库(ARSD)(n = 244)和国际雷特综合征表型数据库(InterRett)(n = 522)获取了766名15岁及以下、基因确诊为雷特综合征女孩的横断面数据。研究了MECP2突变类型与言语语言能力衰退年龄之间的关系,以及衰退前后言语语言能力的水平。ARSD组女性的中位年龄为4.95岁,InterRett组为5.25岁。大多数(89%,685/766)在某个时间点以咿呀学语或单词的形式获得了言语语言能力。在那些获得咿呀学语或单词能力的人中,85%(581/685)经历了这些能力的衰退。携带p.Arg133Cys突变的患者在言语语言能力衰退之前(相对危险度比RRR = 3.45;95%置信区间CI 1.15 - 10.41)和之后(RRR = 5.99;95% CI 2.00 - 17.92)最有可能使用一个或多个单词。患有雷特综合征的女孩在言语和语言的使用以及言语语言能力衰退经历方面存在差异,这些差异部分由基因型解释。

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