Literature DB >> 31069783

A novel WARS mutation (p.Asp314Gly) identified in a Chinese distal hereditary motor neuropathy family.

Binghao Wang1, Xiaobo Li1, Shunxiang Huang1, Huadong Zhao1, Jun Liu2, Zhengmao Hu3, Zhiqiang Lin1, Lei Liu4, Yongzhi Xie1, Qingwen Jin5, Huihui Zhao6, Beisha Tang7, Qi Niu6, Ruxu Zhang1.   

Abstract

Distal hereditary motor neuropathy (dHMN) is a clinically and genetically heterogeneous group of inherited neuropathies characterized by distal limb muscle wasting and weakness with no or minimal sensory abnormalities. To investigate the clinical and genetic features of dHMN caused by WARS mutations in mainland China, we performed Sanger sequencing of the coding and untranslated region (UTR) regions of WARS in 160 unresolved dHMN and Charcot-Marie-Tooth (CMT) index patients. We detected a novel heterozygous variant c.941A>G (p.Asp314Gly) of WARS in an index patient from an autosomal dominant dHMN family including five affected members over three generations. The variant completely co-segregates with the dHMN phenotype in the family, and it was classified as likely pathogenic according to the American College of Medical Genetics and Genomics standards and guidelines. The clinical features included juvenile to adult onset (15-23 years), distal wasting and weakness, minimal sensory disturbance and length-dependent motor axonal degeneration with CMT examination score ranging from 6 to 10. Our report further confirms the role of WARS in dHMN and indicates that the variant c.941A>G (p.Asp314Gly) of WARS is related to a mild to moderate affected and later onset phenotype of dHMN.
© 2019 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd.

Entities:  

Keywords:  zzm321990WARS; distal hereditary motor neuropathy; hTrpRS; novel mutation; phenotype

Mesh:

Substances:

Year:  2019        PMID: 31069783     DOI: 10.1111/cge.13563

Source DB:  PubMed          Journal:  Clin Genet        ISSN: 0009-9163            Impact factor:   4.438


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