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中国吉特曼综合征诊断的分子复杂性分析

Molecular complexity analysis of the diagnosis of Gitelman syndrome in China.

作者信息

Song Wei, Hu Yue, Zhao Ling, Zhang Jinming, Zhang Yu, Wen Jianxuan

机构信息

The Second Affiliated Hospital of Guangzhou University of Chinese Medicine, Guangzhou, Guangdong Province, 510120, China.

Second Clinical College of Guangzhou University of Chinese Medicine, Guangzhou, Guangdong Province, 510120, China.

出版信息

Open Life Sci. 2023 Jun 20;18(1):20220634. doi: 10.1515/biol-2022-0634. eCollection 2023.

Abstract

Gitelman syndrome (GS) is an autosomal recessive renal tubal disease characterized by hypomagnesemia, hypokalemia, and hypocalciuria. The disease is caused by defects in the SLC12A3 gene, which encodes the thiazide diuretic-sensitive sodium chloride cotransporter (NCCT). In this study, a 20-year-old female patient with recurrent hypokalemia was tested for a hypokalemia-related panel using Next Generation Sequencing. Pedigree analysis was performed on her parents (non-consanguineous) and sister using Sanger sequencing. The results revealed that the patient carried compound heterozygous variants of the SLC12A3 gene: c.179C > T (p.T60M) and c.1001G > A (p.R334Q). Furthermore, her asymptomatic 6-year-old sister also carried both mutations. While the p.T60M mutation had been reported previously, the p.R334Q mutation was novel, and amino acid position 334 was identified as a mutation hotspot. Our findings provide an accurate molecular diagnosis that is essential for the diagnosis, counseling, and management of not only the symptomatic patient but also her asymptomatic sister. This study contributes to our understanding of the GS, which has a prevalence of approximately 1 in 40,000 and a heterozygous mutation carrier rate of 1% in Caucasians. Specifically, we observed a compound heterozygous mutation of the SLC12A3 gene in a 20-year-old female patient presenting with clinical symptoms consistent with GS.

摘要

吉特林综合征(GS)是一种常染色体隐性遗传性肾小管疾病,其特征为低镁血症、低钾血症和低钙尿症。该疾病由SLC12A3基因突变引起,该基因编码噻嗪类利尿剂敏感的氯化钠共转运体(NCCT)。在本研究中,一名患有复发性低钾血症的20岁女性患者使用下一代测序技术进行了低钾血症相关检测。使用桑格测序法对其父母(非近亲)和妹妹进行了家系分析。结果显示,该患者携带SLC12A3基因的复合杂合变异:c.179C>T(p.T60M)和c.1001G>A(p.R334Q)。此外,她无症状的6岁妹妹也携带这两种突变。虽然p.T60M突变此前已有报道,但p.R334Q突变是新发现的,并且氨基酸位置334被确定为突变热点。我们的研究结果提供了准确的分子诊断,这不仅对有症状的患者,而且对其无症状的妹妹的诊断、咨询和管理都至关重要。本研究有助于我们对GS的理解,GS在白种人中的患病率约为1/40000,杂合突变携带率为1%。具体而言,我们在一名表现出与GS一致临床症状的20岁女性患者中观察到SLC12A3基因的复合杂合突变。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/2544/10290276/8ce5e519526b/j_biol-2022-0634-fig001.jpg

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