Literature DB >> 24420545

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.

Meena Murthy1, Shengxin Xu, Gianmichele Massimo, Martin Wolley, Richard D Gordon, Michael Stowasser, Kevin M O'Shaughnessy.   

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.

Entities:  

Keywords:  KCNJ5, potassium channel; adrenal cortex; mutation, missense; polymorphism, single nucleotide

Mesh:

Substances:

Year:  2014        PMID: 24420545     DOI: 10.1161/HYPERTENSIONAHA.113.02234

Source DB:  PubMed          Journal:  Hypertension        ISSN: 0194-911X            Impact factor:   10.190


  23 in total

Review 1.  Primary aldosteronism: a common cause of resistant hypertension.

Authors:  Gregory A Kline; Ally P H Prebtani; Alexander A Leung; Ernesto L Schiffrin
Journal:  CMAJ       Date:  2017-06-05       Impact factor: 8.262

Review 2.  Familial hyperaldosteronism type III.

Authors:  S Monticone; M Tetti; J Burrello; F Buffolo; R De Giovanni; F Veglio; T A Williams; P Mulatero
Journal:  J Hum Hypertens       Date:  2017-04-27       Impact factor: 3.012

3.  Channels and pumps in aldosterone-producing adenomas.

Authors:  Celso E Gomez-Sanchez
Journal:  J Clin Endocrinol Metab       Date:  2014-04       Impact factor: 5.958

Review 4.  Aldosterone excess and resistant hypertension: investigation and treatment.

Authors:  Michael Stowasser
Journal:  Curr Hypertens Rep       Date:  2014-07       Impact factor: 5.369

Review 5.  Hyperaldosteronism: How to Discriminate Among Different Disease Forms?

Authors:  Valentina Crudo; Silvia Monticone; Jacopo Burrello; Fabrizio Buffolo; Martina Tetti; Franco Veglio; Paolo Mulatero
Journal:  High Blood Press Cardiovasc Prev       Date:  2016-05-02

6.  A novel somatic mutation 145-147delETEinsK in KCNJ5 increases aldosterone production.

Authors:  F-F Zheng; L-M Zhu; W-L Zhou; Y Zhang; M-Y Li; Y-C Zhu; J-G Wang; D-L Zhu; P-J Gao
Journal:  J Hum Hypertens       Date:  2017-08-24       Impact factor: 3.012

7.  Disordered zonal and cellular CYP11B2 enzyme expression in familial hyperaldosteronism type 3.

Authors:  Celso E Gomez-Sanchez; Xin Qi; Elise P Gomez-Sanchez; Hironobu Sasano; Martin O Bohlen; Max Wisgerhof
Journal:  Mol Cell Endocrinol       Date:  2016-10-25       Impact factor: 4.102

Review 8.  Somatic mutations of the ATP1A1 gene and aldosterone-producing adenomas.

Authors:  Celso E Gomez-Sanchez; Maniselvan Kuppusamy; Elise P Gomez-Sanchez
Journal:  Mol Cell Endocrinol       Date:  2014-12-10       Impact factor: 4.102

9.  Primary aldosteronism: a channelopathy?

Authors:  Celso E Gomez-Sanchez
Journal:  Hypertension       Date:  2014-01-13       Impact factor: 10.190

10.  Mutated KCNJ5 activates the acute and chronic regulatory steps in aldosterone production.

Authors:  Namita G Hattangady; Shigehiro Karashima; Lucy Yuan; Daniela Ponce-Balbuena; José Jalife; Celso E Gomez-Sanchez; Richard J Auchus; William E Rainey; Tobias Else
Journal:  J Mol Endocrinol       Date:  2016-04-20       Impact factor: 5.098

View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.