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在原发性醛固酮增多症的大样本散发病例中,种系突变和罕见编码单核苷酸多态性在 KCNJ5 钾通道中的作用。

Role for germline mutations and a rare coding single nucleotide polymorphism within the KCNJ5 potassium channel in a large cohort of sporadic cases of primary aldosteronism.

机构信息

Clinical Pharmacology Unit, Department of Medicine, Addenbrooke's Hospital, Cambridge, United Kingdom.

出版信息

Hypertension. 2014 Apr;63(4):783-9. doi: 10.1161/HYPERTENSIONAHA.113.02234. Epub 2014 Jan 13.

Abstract

Primary aldosteronism (autonomous aldosterone production with suppressed renin) plays an important pathophysiological role in what has been previously labeled as essential hypertension. Besides the recently described germline mutations in the KCNJ5 potassium channel associated with familial primary aldosteronism, somatic mutations in the same channel have been identified within aldosterone-producing adenomas. In this study, we have resequenced the flanking and coding region of KCNJ5 in peripheral blood DNA from 251 white subjects with primary aldosteronism to look for rare variants that might be important for the pathophysiology of sporadic primary aldosteronism. We have identified 3 heterozygous missense mutations (R52H, E246K, and G247R) in the cohort and found that 12 (5% of the cohort) were carriers for the rare nonsynonymous single nucleotide polymorphism rs7102584 causing E282Q substitution of KCNJ5. By expressing the channels in Xenopus oocytes and human adrenal H295R cells, we have shown that the R52H, E246K, and E282Q substitutions are functional, but the G247R mutation is indistinguishable from wild type. Although the functional substitutions are remote from the selectivity filter, they affect the inward-rectification, the ability of the KCNJ5 channels to conduct Na(+) currents and ATII-induced aldosterone release from the H295R cell line. Together these data suggest that germline variation in the KCNJ5 gene has a role to play in the common sporadic form as well as the much rarer syndromic forms of primary aldosteronism.

摘要

原发性醛固酮增多症(自主醛固酮分泌,肾素受抑制)在以前被归类为原发性高血压中具有重要的病理生理作用。除了最近描述的与家族性原发性醛固酮增多症相关的 KCNJ5 钾通道种系突变外,在醛固酮分泌腺瘤中也发现了同一通道的体细胞突变。在这项研究中,我们对 251 例原发性醛固酮增多症白人患者的外周血 DNA 进行了 KCNJ5 侧翼和编码区的重测序,以寻找可能对散发性原发性醛固酮增多症病理生理学重要的稀有变异。我们在队列中发现了 3 个杂合错义突变(R52H、E246K 和 G247R),并发现 12 例(队列的 5%)为罕见非同义单核苷酸多态性 rs7102584 的携带者,该多态性导致 KCNJ5 的 E282Q 取代。通过在非洲爪蟾卵母细胞和人肾上腺 H295R 细胞中表达这些通道,我们表明 R52H、E246K 和 E282Q 取代是功能性的,但 G247R 突变与野生型无法区分。尽管功能取代远离选择性过滤器,但它们会影响 KCNJ5 通道的内向整流、Na(+)电流的传导能力以及来自 H295R 细胞系的 ATII 诱导的醛固酮释放。这些数据表明,KCNJ5 基因的种系变异在常见的散发性形式以及更为罕见的综合征形式的原发性醛固酮增多症中都具有作用。

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