| Literature DB >> 30617279 |
Nannan Li1, Ling Wang1, Xiaoyi Sun1, Zhongjiao Lu1, Xueling Suo2, Junying Li1, Jiaxin Peng1, Rong Peng3.
Abstract
Distal spinal muscular atrophy (dSMA) is a rare clinically and genetically heterogeneous group of inherited disorders characterized by progressive distal muscle weakness and wasting. So far, more than 65% of patients with dSMA have undiscovered genetic mutations. Recently, compound heterozygous mutations in the vaccinia-related kinase 1 (VRK1) gene have been identified for the first time in two siblings with adult-onset dSMA from an Ashkenazi Jewish family. Here, we also report two affected siblings with adult-onset dSMA in a Chinese family. Whole exome sequencing and subsequent Sanger sequencing identified a novel nonsense mutation (c.1124G >A, p.W375*) in exon 12 of the VRK1 gene, co-segregating with the dSMA phenotype in an autosomal recessive pattern. In conclusion, our findings identify a novel nonsense mutation p.W375* in the VRK1 gene in a Chinese family with autosomal recessive dSMA and broaden the genetic spectrum of VRK1-associated dSMA.Entities:
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Year: 2019 PMID: 30617279 DOI: 10.1038/s10038-018-0553-5
Source DB: PubMed Journal: J Hum Genet ISSN: 1434-5161 Impact factor: 3.172