Literature DB >> 30617279

A novel mutation in VRK1 associated with distal spinal muscular atrophy.

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.

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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


  6 in total

1.  Dysregulation of Cellular VRK1, BAF, and Innate Immune Signaling by the Vaccinia Virus B12 Pseudokinase.

Authors:  Alexandria C Linville; Amber B Rico; Helena Teague; Lucy E Binsted; Geoffrey L Smith; Jonas D Albarnaz; Matthew S Wiebe
Journal:  J Virol       Date:  2022-05-11       Impact factor: 6.549

2.  Multivalent DNA and nucleosome acidic patch interactions specify VRK1 mitotic localization and activity.

Authors:  Gabrielle R Budziszewski; Yani Zhao; Cathy J Spangler; Katarzyna M Kedziora; Michael R Williams; Dalal N Azzam; Aleksandra Skrajna; Yuka Koyama; Andrew P Cesmat; Holly C Simmons; Eyla C Arteaga; Joshua D Strauss; Dmitri Kireev; Robert K McGinty
Journal:  Nucleic Acids Res       Date:  2022-05-06       Impact factor: 19.160

3.  VRK1 (Y213H) homozygous mutant impairs Cajal bodies in a hereditary case of distal motor neuropathy.

Authors:  Ana T Marcos; Elena Martín-Doncel; Patricia Morejón-García; Iñigo Marcos-Alcalde; Paulino Gómez-Puertas; María Segura-Puimedon; Lluis Armengol; José M Navarro-Pando; Pedro A Lazo
Journal:  Ann Clin Transl Neurol       Date:  2020-05-04       Impact factor: 4.511

4.  Motor neuron diseases caused by a novel VRK1 variant - A genotype/phenotype study.

Authors:  Maryam Sedghi; Ali-Reza Moslemi; Montse Olive; Masoud Etemadifar; Behnaz Ansari; Jafar Nasiri; Leila Emrahi; Hamid-Reza Mianesaz; Nigel G Laing; Homa Tajsharghi
Journal:  Ann Clin Transl Neurol       Date:  2019-09-27       Impact factor: 4.511

5.  Dysfunctional Homozygous VRK1-D263G Variant Impairs the Assembly of Cajal Bodies and DNA Damage Response in Hereditary Spastic Paraplegia.

Authors:  Patricia Morejon-Garcia; Boris Keren; Iñigo Marcos-Alcalde; Paulino Gomez-Puertas; Fanny Mochel; Pedro A Lazo
Journal:  Neurol Genet       Date:  2021-09-02

6.  Adult-Onset Spinal Muscular Atrophy due to Mutations in the VRK1 Gene.

Authors:  Angela Sung; Paolo Moretti; Aziz Shaibani
Journal:  Neurol Genet       Date:  2021-06-22
  6 in total

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