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病例报告:SCN4A基因p.R1135H变异与甲状腺毒症合并导致低钾性周期性麻痹。

Case report: SCN4A p.R1135H gene variant in combination with thyrotoxicosis causing hypokalemic periodic paralysis.

作者信息

Zhang Zhi, Xiao Banghui

机构信息

Department of Endocrinology and Metabolism, Affiliated Hospital of Guizhou Medical University, Guiyang, China.

出版信息

Front Neurol. 2023 Jan 17;13:1078784. doi: 10.3389/fneur.2022.1078784. eCollection 2022.

Abstract

Hypokalemic periodic paralysis (HPP) is a heterogeneous group of diseases characterized by intermittent episodes of delayed paralysis of skeletal muscle with episodes of hypokalemia, caused by variants in CACNA1S or SCN4A genes, or secondary to thyrotoxicosis, Sjogren syndrome, primary aldosteronism, etc. HPP may be the only presentation in Andersen-Tawil syndrome in which the majority of cases are caused by pathogenic variants in the KCNJ2 gene. We present a case of a 29-year-old male with hypokalemic periodic paralysis. The patient began to experience recurrent weakness of the extremities at the age of 26, which was effectively treated with potassium supplementation. He had recently developed dry mouth, palpitations, weight loss, and even dyspnea, with a serum potassium level as low as 1.59 mmol/L. The results of auxiliary examinations showed Graves' disease, and genetic testing indicated a missense variant, NM_000334.4 (SCN4A):c.3404G>A (p.R1135H). He did not experience periodic paralysis during follow-up after lifestyle guidance and treatment of thyrotoxicosis with radioactive iodine. It is a rare case of SCN4A p.R1135H gene variant combined with hyperthyroidism resulting in HPP with respiratory muscle paralysis to raise awareness of the disease and avoid misdiagnosis and missed diagnosis.

摘要

低钾性周期性麻痹(HPP)是一组异质性疾病,其特征为骨骼肌延迟性麻痹的间歇性发作,并伴有低钾血症发作,由CACNA1S或SCN4A基因变异引起,或继发于甲状腺毒症、干燥综合征、原发性醛固酮增多症等。HPP可能是安德森-塔维尔综合征的唯一表现,其中大多数病例由KCNJ2基因的致病变异引起。我们报告一例29岁男性低钾性周期性麻痹病例。患者26岁时开始反复出现四肢无力,补充钾后得到有效治疗。他最近出现口干、心悸、体重减轻,甚至呼吸困难,血清钾水平低至1.59 mmol/L。辅助检查结果显示为格雷夫斯病,基因检测表明存在一个错义变异,NM_000334.4(SCN4A):c.3404G>A(p.R1135H)。在进行生活方式指导和放射性碘治疗甲状腺毒症后随访期间,他未再出现周期性麻痹。这是一例罕见的SCN4A p.R1135H基因变异合并甲状腺功能亢进导致HPP并伴有呼吸肌麻痹的病例,以提高对该疾病的认识,避免误诊和漏诊。

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本文引用的文献

2
Treatment Updates for Neuromuscular Channelopathies.
Curr Treat Options Neurol. 2020;22(10):34. doi: 10.1007/s11940-020-00644-2. Epub 2020 Aug 22.
3
Contribution of Asian Haplotype of KCNJ18 to Susceptibility to and Ethnic Differences in Thyrotoxic Periodic Paralysis.
J Clin Endocrinol Metab. 2019 Dec 1;104(12):6338-6344. doi: 10.1210/jc.2019-00672.
4
Skeletal Muscle Channelopathies.
Neurotherapeutics. 2018 Oct;15(4):954-965. doi: 10.1007/s13311-018-00678-0.
5
Dichlorphenamide: A Review in Primary Periodic Paralyses.
Drugs. 2016 Mar;76(4):501-7. doi: 10.1007/s40265-016-0559-2.
6
Randomized, placebo-controlled trials of dichlorphenamide in periodic paralysis.
Neurology. 2016 Apr 12;86(15):1408-1416. doi: 10.1212/WNL.0000000000002416. Epub 2016 Feb 10.
7
Voltage-Gated Sodium Channels: Structure, Function, Pharmacology, and Clinical Indications.
J Med Chem. 2015 Sep 24;58(18):7093-118. doi: 10.1021/jm501981g. Epub 2015 May 14.
8
Evaluating the efficacy of primary treatment for graves' disease complicated by thyrotoxic periodic paralysis.
Int J Endocrinol. 2014;2014:949068. doi: 10.1155/2014/949068. Epub 2014 Aug 3.
9
NaV1.4 mutations cause hypokalaemic periodic paralysis by disrupting IIIS4 movement during recovery.
Brain. 2014 Apr;137(Pt 4):998-1008. doi: 10.1093/brain/awu015. Epub 2014 Feb 18.
10
Prevalence study of genetically defined skeletal muscle channelopathies in England.
Neurology. 2013 Apr 16;80(16):1472-5. doi: 10.1212/WNL.0b013e31828cf8d0. Epub 2013 Mar 20.

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