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先天性肌强直和周期性低钾血症性麻痹在一个血缘婚姻家系中:一种新型 CLCN1 突变和 SCN4A 突变共存。

Myotonia congenita and periodic hypokalemia paralysis in a consanguineous marriage pedigree: Coexistence of a novel CLCN1 mutation and an SCN4A mutation.

机构信息

Department of Medical Genetics, The Second Xiangya Hospital, Central South University, Changsha, Hunan, China.

Department of Gastroenterology, The Second Xiangya Hospital, Central South University, Changsha, Hunan, China.

出版信息

PLoS One. 2020 May 14;15(5):e0233017. doi: 10.1371/journal.pone.0233017. eCollection 2020.

Abstract

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.

摘要

先天性肌强直和低钾周期性麻痹 2 型都是由电压门控氯离子通道 CLCN1 基因编码的电压门控氯离子通道 CLC-1 和电压门控钠离子通道 Nav1.4 基因编码的 SCN4A 基因突变引起的罕见遗传性通道病。同时存在这两个基因的突变的患者表现出不同于这些基因单独突变的独特症状。在这里,我们描述了一个在近亲结婚家系中患有肌强直和周期性麻痹的患者。通过全外显子组测序,在家系中发现了 CLCN1 基因中的新型 F306S 变异和 SCN4A 基因中的已知 R222W 突变。膜片钳分析表明,F306S 突变降低了 CLC-1 的开放概率和氯离子电导。我们的研究扩展了 CLCN1 突变数据库。我们强调了全外显子组测序在非典型肌强直患者中的鉴别诊断价值。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f4d7/7224471/8522fb6a383b/pone.0233017.g001.jpg

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