Literature DB >> 28544625

A mutation in GABRB3 associated with Dravet syndrome.

Sy Vinh Le1, Phan Hoang Truc Le2, Thi Khanh Van Le3, Thi Thuy Kieu Huynh3, Thi Thu Hang Do4.   

Abstract

Dravet syndrome is a rare and severe type of epilepsy in infants. Approximately, 70-80% of patients with Dravet syndrome have mutations in SCN1A, the gene encoding the alpha-1 subunit of the sodium channel, while some simplex patients have variants in one of several other genes, including but not limited to GABRA1, SCN2A, STXBP1, GABRG2, and SCN1B. In this study, we performed exome sequencing in six patients with SCN1A-negative Dravet syndrome to identify other genes related to this disorder. In one affected individual, we detected a novel de novo heterozygous missense variant, c.695G>A, p.(Arg232Gln), in GABRB3, the gene encoding the β3-subunit of the gamma-aminobutyric acid type A (GABAA) receptor, which mediates inhibitory signaling within the central nervous system. In summary, the data in this study identify GABRB3 as a candidate gene for Dravet syndrome.
© 2017 Wiley Periodicals, Inc.

Entities:  

Keywords:  Dravet syndrome; GABRB3; SCN1A; exome sequencing

Mesh:

Substances:

Year:  2017        PMID: 28544625     DOI: 10.1002/ajmg.a.38282

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  9 in total

1.  Synaptic clustering differences due to different GABRB3 mutations cause variable epilepsy syndromes.

Authors:  Yi-Wu Shi; Qi Zhang; Kefu Cai; Sarah Poliquin; Wangzhen Shen; Nathan Winters; Yong-Hong Yi; Jie Wang; Ningning Hu; Robert L Macdonald; Wei-Ping Liao; Jing-Qiong Kang
Journal:  Brain       Date:  2019-10-01       Impact factor: 13.501

2.  Altered inhibitory synapses in de novo GABRA5 and GABRA1 mutations associated with early onset epileptic encephalopathies.

Authors:  Ciria C Hernandez; Wenshu XiangWei; Ningning Hu; Dingding Shen; Wangzhen Shen; Andre H Lagrange; Yujia Zhang; Lifang Dai; Changhong Ding; Zhaohui Sun; Jiasheng Hu; Hongmin Zhu; Yuwu Jiang; Robert L Macdonald
Journal:  Brain       Date:  2019-07-01       Impact factor: 13.501

3.  SCN1B and SCN2B gene variants analysis in dravet syndrome patients: Analysis of 22 cases.

Authors:  Jiao-E Gong; Hong-Mei Liao; Hong-Yu Long; Xiang-Min Li; Li-Li Long; Luo Zhou; Wen-Ping Gu; Shao-Hua Lu; Qiang Qu; Li-Min Yang; Bo Xiao; Jian Qu
Journal:  Medicine (Baltimore)       Date:  2019-03       Impact factor: 1.889

Review 4.  A novel GABRB3 variant in Dravet syndrome: Case report and literature review.

Authors:  Piero Pavone; Xena Giada Pappalardo; Simona D Marino; Laura Sciuto; Giovanni Corsello; Martino Ruggieri; Enrico Parano; Maria Piccione; Raffaele Falsaperla
Journal:  Mol Genet Genomic Med       Date:  2020-09-18       Impact factor: 2.183

5.  Phenotype Expression Variability in Children with GABRB3 Heterozygous Mutations.

Authors:  Abdulhafeez M Khair; Alana E Salvucci
Journal:  Oman Med J       Date:  2021-03-31

6.  Do All Roads Lead to Rome? Genes Causing Dravet Syndrome and Dravet Syndrome-Like Phenotypes.

Authors:  Jiangwei Ding; Lei Wang; Zhe Jin; Yuanyuan Qiang; Wenchao Li; Yangyang Wang; Changliang Zhu; Shucai Jiang; Lifei Xiao; Xiaoyan Hao; Xulei Hu; Xinxiao Li; Feng Wang; Tao Sun
Journal:  Front Neurol       Date:  2022-03-11       Impact factor: 4.003

Review 7.  Variable Expression of GABAA Receptor Subunit Gamma 2 Mutation in a Nuclear Family Displaying Developmental and Encephalopathic Phenotype.

Authors:  Gerald Nwosu; Shilpa B Reddy; Heather Rose Mead Riordan; Jing-Qiong Kang
Journal:  Int J Mol Sci       Date:  2022-08-26       Impact factor: 6.208

8.  The K328M substitution in the human GABAA receptor gamma2 subunit causes GEFS+ and premature sudden death in knock-in mice.

Authors:  Shimian Qu; Chengwen Zhou; Rachel Howe; Wangzhen Shen; Xuan Huang; Mackenzie Catron; Ningning Hu; Robert L Macdonald
Journal:  Neurobiol Dis       Date:  2021-02-11       Impact factor: 5.996

9.  Heterozygous deletion of SCN2A and SCN3A in a patient with autism spectrum disorder and Tourette syndrome: a case report.

Authors:  Kathrin Nickel; Ludger Tebartz van Elst; Katharina Domschke; Birgitta Gläser; Friedrich Stock; Dominique Endres; Simon Maier; Andreas Riedel
Journal:  BMC Psychiatry       Date:  2018-08-02       Impact factor: 3.630

  9 in total

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