Unité de Génétique Médicale et laboratoire associé INSERM à l'Unité UMR_S910, Université Saint-Joseph, Beirut, Lebanon.
J Intellect Disabil Res. 2012 Apr;56(4):415-20. doi: 10.1111/j.1365-2788.2011.01479.x. Epub 2011 Sep 29.
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.
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.
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.
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.
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。
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。
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。
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。