| Literature DB >> 27335120 |
J Christopher Hennings1, Olga Andrini2, Nicolas Picard2, Marc Paulais2, Antje K Huebner1, Irma Karen Lopez Cayuqueo3,4, Yohan Bignon2, Mathilde Keck2, Nicolas Cornière5, David Böhm1, Thomas J Jentsch6, Régine Chambrey3,7,8, Jacques Teulon9, Christian A Hübner1, Dominique Eladari10,7,11.
Abstract
Chloride transport by the renal tubule is critical for blood pressure (BP), acid-base, and potassium homeostasis. Chloride uptake from the urinary fluid is mediated by various apical transporters, whereas basolateral chloride exit is thought to be mediated by ClC-Ka/K1 and ClC-Kb/K2, two chloride channels from the ClC family, or by KCl cotransporters from the SLC12 gene family. Nevertheless, the localization and role of ClC-K channels is not fully resolved. Because inactivating mutations in ClC-Kb/K2 cause Bartter syndrome, a disease that mimics the effects of the loop diuretic furosemide, ClC-Kb/K2 is assumed to have a critical role in salt handling by the thick ascending limb. To dissect the role of this channel in detail, we generated a mouse model with a targeted disruption of the murine ortholog ClC-K2. Mutant mice developed a Bartter syndrome phenotype, characterized by renal salt loss, marked hypokalemia, and metabolic alkalosis. Patch-clamp analysis of tubules isolated from knockout (KO) mice suggested that ClC-K2 is the main basolateral chloride channel in the thick ascending limb and in the aldosterone-sensitive distal nephron. Accordingly, ClC-K2 KO mice did not exhibit the natriuretic response to furosemide and exhibited a severely blunted response to thiazide. We conclude that ClC-Kb/K2 is critical for salt absorption not only by the thick ascending limb, but also by the distal convoluted tubule.Entities:
Keywords: Bartter-s syndrome; chloride channels; ion transport; transgenic mouse
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Year: 2016 PMID: 27335120 PMCID: PMC5198284 DOI: 10.1681/ASN.2016010085
Source DB: PubMed Journal: J Am Soc Nephrol ISSN: 1046-6673 Impact factor: 10.121