• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

对怀疑患有雷特综合征的黎巴嫩患者进行 MECP2 基因的分子筛查:一例伴有新型缺失突变的轻度病例报告。

Molecular screening of MECP2 gene in a cohort of Lebanese patients suspected with Rett syndrome: report on a mild case with a novel indel mutation.

机构信息

Unité de Génétique Médicale et laboratoire associé INSERM à l'Unité UMR_S910, Université Saint-Joseph, Beirut, Lebanon.

出版信息

J Intellect Disabil Res. 2012 Apr;56(4):415-20. doi: 10.1111/j.1365-2788.2011.01479.x. Epub 2011 Sep 29.

DOI:10.1111/j.1365-2788.2011.01479.x
PMID:21954873
Abstract

BACKGROUND

Rett syndrome (RTT), an X-linked, dominant, neurodevelopment disorder represents 10% of female subjects with profound intellectual disability. Mutations in the MECP2 gene are responsible for up to 95% of the classical RTT cases, and nearly 500 different mutations distributed throughout the gene have been reported.

METHODS

We report here the molecular study of two isoforms, MECP2_e1 and MECP2_e2, in 45 Lebanese girls presenting developmental delay and at least one of the following features: microcephaly, neurodegeneration, abnormal behaviour, stereotypical hand movements, teeth grinding and difficulty in walking. Mutation screening was performed by denaturating high-performance liquid chromatography combined with direct sequencing.

RESULTS

Sixteen variants were noted, of which 14 have been previously reported: five suspected polymorphisms and nine mutations. Two variants were novel mutations in exon 4: c.1093_1095delGAG (p.E365del) and c.1164_1184delACCTCCACCTGAGCCCGAGAGinsCTGAGCCCCAGGACTTGAGCA (p.P388PfsX389). The deletion was found in an 8-year-old girl with typical clinical features of RTT. The indel was found in a 6-year-old girl with a very mild phenotype.

CONCLUSION

Genotype/phenotype correlation is discussed and the importance of a molecular study of MECP2 gene in patients with very mild features or a regression after the age of 2 is raised.

摘要

背景

雷特综合征(RTT)是一种 X 连锁显性、神经发育障碍,占女性严重智力障碍患者的 10%。MECP2 基因突变负责高达 95%的经典 RTT 病例,并且已经报道了近 500 种分布在整个基因中的不同突变。

方法

我们在此报告对 45 名黎巴嫩女孩的两个同工型 MECP2_e1 和 MECP2_e2 的分子研究,这些女孩表现出发育迟缓,至少具有以下一种特征:小头畸形、神经退行性变、异常行为、刻板手运动、磨牙和行走困难。通过变性高效液相色谱法结合直接测序进行突变筛查。

结果

共发现 16 种变体,其中 14 种已被先前报道:5 种疑似多态性和 9 种突变。两种变体是外显子 4 中的新突变:c.1093_1095delGAG(p.E365del)和 c.1164_1184delACCTCCACCTGAGCCCGAGAGinsCTGAGCCCCAGGACTTGAGCA(p.P388PfsX389)。缺失在一名 8 岁女孩中被发现,其具有典型的 RTT 临床特征。插入缺失在一名 6 岁女孩中被发现,其表型非常轻微。

结论

讨论了基因型/表型相关性,并提出了在具有非常轻微特征或 2 岁后出现退行性变的患者中对 MECP2 基因进行分子研究的重要性。

相似文献

1
Molecular screening of MECP2 gene in a cohort of Lebanese patients suspected with Rett syndrome: report on a mild case with a novel indel mutation.对怀疑患有雷特综合征的黎巴嫩患者进行 MECP2 基因的分子筛查:一例伴有新型缺失突变的轻度病例报告。
J Intellect Disabil Res. 2012 Apr;56(4):415-20. doi: 10.1111/j.1365-2788.2011.01479.x. Epub 2011 Sep 29.
2
MECP2 mutations in Serbian Rett syndrome patients.塞尔维亚雷特综合征患者中的MECP2基因突变
Acta Neurol Scand. 2007 Dec;116(6):413-9. doi: 10.1111/j.1600-0404.2007.00893.x.
3
Spectrum of MECP2 mutations in New Zealand Rett syndrome patients.新西兰雷特综合征患者中MECP2基因突变谱。
N Z Med J. 2009 Jun 5;122(1296):21-8.
4
Phenotypic and genotypic variability in four males with MECP2 gene sequence aberrations including a novel deletion.四名男性 MECP2 基因序列异常患者(包括一例新的缺失)的表型和基因型变异性。
Pediatr Res. 2010 May;67(5):551-6. doi: 10.1203/PDR.0b013e3181d4ecf7.
5
Clinical profiles of four patients with Rett syndrome carrying a novel exon 1 mutation or genomic rearrangement in the MECP2 gene.四名患有雷特综合征的患者的临床特征,这些患者在MECP2基因中携带一种新的外显子1突变或基因组重排。
Clin Genet. 2006 Apr;69(4):319-26. doi: 10.1111/j.1399-0004.2006.00604.x.
6
MECP2 mutations and clinical correlations in Greek children with Rett syndrome and associated neurodevelopmental disorders.希腊患有雷特综合征及相关神经发育障碍儿童的MECP2突变与临床相关性
Brain Dev. 2012 Jun;34(6):487-95. doi: 10.1016/j.braindev.2011.09.002. Epub 2011 Oct 6.
7
MECP2 mutations in Czech patients with Rett syndrome and Rett-like phenotypes: novel mutations, genotype-phenotype correlations and validation of high-resolution melting analysis for mutation scanning.捷克雷特综合征及类雷特综合征表型患者的MECP2突变:新突变、基因型-表型相关性及用于突变扫描的高分辨率熔解分析的验证
J Hum Genet. 2016 Jul;61(7):617-25. doi: 10.1038/jhg.2016.19. Epub 2016 Mar 17.
8
X chromosome inactivation in Rett Syndrome and its correlations with MECP2 mutations and phenotype.雷特综合征中的X染色体失活及其与MECP2突变和表型的相关性。
J Child Neurol. 2008 Jan;23(1):22-5. doi: 10.1177/0883073807307077.
9
Social impairments in Rett syndrome: characteristics and relationship with clinical severity.雷特综合征的社交障碍:特征及与临床严重程度的关系。
J Intellect Disabil Res. 2012 Mar;56(3):233-47. doi: 10.1111/j.1365-2788.2011.01404.x. Epub 2011 Mar 8.
10
Deleterious mutations in exon 1 of MECP2 in Rett syndrome.雷特综合征中MECP2基因第1外显子的有害突变。
Eur J Med Genet. 2006 Jul-Aug;49(4):313-22. doi: 10.1016/j.ejmg.2005.11.002. Epub 2005 Dec 20.

引用本文的文献

1
Oral health experiences of individuals with Rett syndrome: a retrospective study.雷特综合征患者的口腔健康体验:一项回顾性研究。
BMC Oral Health. 2018 Nov 29;18(1):195. doi: 10.1186/s12903-018-0651-y.
2
A review of the diverse genetic disorders in the Lebanese population: highlighting the urgency for community genetic services.黎巴嫩人群中各种遗传疾病的综述:强调社区遗传服务的紧迫性。
J Community Genet. 2015 Jan;6(1):83-105. doi: 10.1007/s12687-014-0203-3. Epub 2014 Sep 27.
3
Brief report: MECP2 mutations in people without Rett syndrome.
简短报告:无雷特综合征患者中的MECP2基因突变
J Autism Dev Disord. 2014 Mar;44(3):703-11. doi: 10.1007/s10803-013-1902-z.