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与严重且进行性婴儿期痉挛性麻痹相关的ALS2基因新突变。

A novel mutation in ALS2 associated with severe and progressive infantile onset of spastic paralysis.

作者信息

Tariq Huma, Mukhtar Shahid, Naz Sadaf

机构信息

a School of Biological Sciences , University of the Punjab , Lahore , Pakistan.

b Punjab Institute of Neurosciences , Lahore General Hospital , Lahore , Pakistan.

出版信息

J Neurogenet. 2017 Mar-Jun;31(1-2):26-29. doi: 10.1080/01677063.2017.1324441. Epub 2017 May 13.

DOI:10.1080/01677063.2017.1324441
PMID:28502191
Abstract

Infantile onset ascending spastic paralysis (IAHSP) is a type of recessively inherited spastic paraplegia. We investigated the clinical and genetic cause of a recessively inherited disorder in two siblings manifesting severe spasticity in the lower limbs which hindered their gait. A novel homozygous nonsense mutation c.1918 C > T (p.Arg640*) was identified after whole-exome sequencing within ALS2 in the DNA of both patients. The obligate carriers were heterozygous for the mutation and other unaffected members were homozygous for the wild type allele. The variant was absent from 100 control chromosomes and all public databases. This report extends the allelic heterogeneity of ALS2 mutations and emphasizes the importance of genetic testing for diagnosis of pediatric disorders.

摘要

婴儿期起病的上行性痉挛性麻痹(IAHSP)是一种隐性遗传性痉挛性截瘫。我们对两名表现为下肢严重痉挛从而影响步态的同胞所患隐性遗传疾病的临床和遗传病因进行了研究。通过全外显子组测序,在两名患者的DNA中发现了位于ALS2基因内的一个新的纯合无义突变c.1918 C>T(p.Arg640*)。必然携带者为该突变的杂合子,其他未受影响的成员为野生型等位基因的纯合子。在100条对照染色体和所有公共数据库中均未发现该变异。本报告扩展了ALS2突变的等位基因异质性,并强调了基因检测对儿科疾病诊断的重要性。

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