Myotonia congenita and hypokalemic periodic paralysis type 2 are both rare genetic channelopathies caused by mutations in the CLCN1 gene encoding voltage-gated chloride channel CLC-1 and the SCN4A gene encoding voltage-gated sodium channel Nav1.4. The patients with concomitant mutations in both genes manifested different unique symptoms from mutations in these genes separately. Here, we describe a patient with myotonia and periodic paralysis in a consanguineous marriage pedigree. By using whole-exome sequencing, a novel F306S variant in the CLCN1 gene and a known R222W mutation in the SCN4A gene were identified in the pedigree. Patch clamp analysis revealed that the F306S mutant reduced the opening probability of CLC-1 and chloride conductance. Our study expanded the CLCN1 mutation database. We emphasized the value of whole-exome sequencing for differential diagnosis in atypical myotonic patients.
Myotonia congenita and periodic hypokalemia paralysis in a consanguineous marriage pedigree: Coexistence of a novel CLCN1 mutation and an SCN4A mutation.
近亲结婚家系中的先天性肌强直和周期性低钾血症麻痹:一种新的 CLCN1 突变和一种 SCN4A 突变的共存
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作者:Zhao Chenyu, Tang DongFang, Huang Hui, Tang Haiyan, Yang Yuan, Yang Min, Luo Yingying, Tao Huai, Tang Jianguang, Zhou Xi, Shi Xiaoliu
| 期刊: | PLoS One | 影响因子: | 2.600 |
| 时间: | 2020 | 起止号: | 2020 May 14; 15(5):e0233017 |
| doi: | 10.1371/journal.pone.0233017 | 研究方向: | 其它 |
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