Literature DB >> 29441481

Whole-exome sequencing reveals a rare interferon gamma receptor 1 mutation associated with myasthenia gravis.

Guoyan Qi1, Peng Liu2, Shanshan Gu2, Hongxia Yang2, Huimin Dong2, Yinping Xue2.   

Abstract

Our study is aimed to explore the underlying genetic basis of myasthenia gravis. We collected a Chinese pedigree with myasthenia gravis, and whole-exome sequencing was performed on the two affected siblings and their parents. The candidate pathogenic gene was identified by bioinformatics filtering, which was further verified by Sanger sequencing. The homozygous mutation c.G40A (p.V14M) in interferon gamma receptor 1was identified. Moreover, the mutation was also detected in 3 cases of 44 sporadic myasthenia gravis patients. The p.V14M substitution in interferon gamma receptor 1 may affect the signal peptide function and the translocation on cell membrane, which could disrupt the binding of the ligand of interferon gamma and antibody production, contributing to myasthenia gravis susceptibility. We discovered that a rare variant c.G40A in interferon gamma receptor 1 potentially contributes to the myasthenia gravis pathogenesis. Further functional studies are needed to confirm the effect of the interferon gamma receptor 1 on the myasthenia gravis phenotype.

Entities:  

Keywords:  Autoimmune disease; Interferon gamma receptor 1; Myasthenia gravis; Pathologenesis; Whole-exome sequencing

Mesh:

Substances:

Year:  2018        PMID: 29441481     DOI: 10.1007/s10072-018-3275-8

Source DB:  PubMed          Journal:  Neurol Sci        ISSN: 1590-1874            Impact factor:   3.307


  36 in total

1.  Identification of deleterious mutations within three human genomes.

Authors:  Sung Chun; Justin C Fay
Journal:  Genome Res       Date:  2009-07-14       Impact factor: 9.043

2.  Role for interferon-gamma in rat strains with different susceptibility to experimental autoimmune myasthenia gravis.

Authors:  H B Wang; F D Shi; H Li; P H van der Meide; H G Ljunggren; H Link
Journal:  Clin Immunol       Date:  2000-05       Impact factor: 3.969

3.  MutationTaster2: mutation prediction for the deep-sequencing age.

Authors:  Jana Marie Schwarz; David N Cooper; Markus Schuelke; Dominik Seelow
Journal:  Nat Methods       Date:  2014-04       Impact factor: 28.547

4.  Decreased mRNA expression of TNF-alpha and IL-10 in non-stimulated peripheral blood mononuclear cells in myasthenia gravis.

Authors:  W X Huang; P Huang; S Fredrikson; R Pirskanen; J Hillert
Journal:  Eur J Neurol       Date:  2000-03       Impact factor: 6.089

5.  Cytokines and the pathogenesis of myasthenia gravis.

Authors:  G X Zhang; V Navikas; H Link
Journal:  Muscle Nerve       Date:  1997-05       Impact factor: 3.217

6.  Crystal structure of a complex between interferon-gamma and its soluble high-affinity receptor.

Authors:  M R Walter; W T Windsor; T L Nagabhushan; D J Lundell; C A Lunn; P J Zauodny; S K Narula
Journal:  Nature       Date:  1995-07-20       Impact factor: 49.962

7.  Myasthenia gravis occurring in families.

Authors:  C Herrmann
Journal:  Neurology       Date:  1966-01       Impact factor: 9.910

8.  Exome sequencing detection of two untranslated GFPT1 mutations in a family with limb-girdle myasthenia.

Authors:  R A Maselli; J Arredondo; J Nguyen; M Lara; F Ng; M Ngo; J M Pham; Q Yi; J M Stajich; K McDonald; M A Hauser; R L Wollmann
Journal:  Clin Genet       Date:  2013-03-11       Impact factor: 4.438

9.  MYASTHENIA GRAVIS IN TWO SIBLINGS.

Authors:  G G CELESIA
Journal:  Arch Neurol       Date:  1965-02

10.  Interaction between the components of the interferon gamma receptor complex.

Authors:  S V Kotenko; L S Izotova; B P Pollack; T M Mariano; R J Donnelly; G Muthukumaran; J R Cook; G Garotta; O Silvennoinen; J N Ihle
Journal:  J Biol Chem       Date:  1995-09-08       Impact factor: 5.157

View more
  1 in total

1.  A Novel missense mutation of COL2A1 gene in a large family with stickler syndrome type I.

Authors:  Xiuzhen Liu; Hongliang Dong; Yuerong Gong; Lianqing Wang; Ruyi Zhang; Tihua Zheng; Yuxi Zheng; Shuang Shen; Chelsea Zheng; Mingming Tian; Naiguo Liu; Xiaolin Zhang; Qing Yin Zheng
Journal:  J Cell Mol Med       Date:  2022-01-21       Impact factor: 5.310

  1 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.