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钾离子通道ROMK突变揭示的巴特综合征的遗传异质性。

Genetic heterogeneity of Bartter's syndrome revealed by mutations in the K+ channel, ROMK.

作者信息

Simon D B, Karet F E, Rodriguez-Soriano J, Hamdan J H, DiPietro A, Trachtman H, Sanjad S A, Lifton R P

机构信息

Howard Hughes Medical Institute, Department of Medicine, Boyer Center for Molecular Medicine, Yale University School of Medicine, New Haven, Connecticut 06510, USA.

出版信息

Nat Genet. 1996 Oct;14(2):152-6. doi: 10.1038/ng1096-152.

Abstract

Mutations in the Na-K-2Cl cotransporter (NKCC2), a mediator of renal salt reabsorption, cause Bartter's syndrome, featuring salt wasting, hypokalaemic alkalosis, hypercalciuria and low blood pressure. NKCC2 mutations can be excluded in some Bartter's kindreds, prompting examination of regulators of cotransporter activity. One regulator is believed to be ROMK, an ATP-sensitive K+ channel that 'recycles' reabsorbed K+ back to the tubule lumen. Examination of the ROMK gene reveals mutations that co-segregate with the disease and disrupt ROMK function in four Bartter's kindreds. Our findings establish the genetic heterogeneity of Bartter's syndrome, and demonstrate the physiologic role of ROMK in vivo.

摘要

钠-钾-2氯协同转运蛋白(NKCC2)是肾脏盐重吸收的介质,其突变会导致巴特综合征,表现为盐消耗、低钾性碱中毒、高钙尿症和低血压。在一些巴特综合征家族中可排除NKCC2突变,这促使人们对协同转运蛋白活性的调节因子进行研究。一种调节因子被认为是ROMK,它是一种ATP敏感性钾通道,可将重吸收的钾“循环”回肾小管管腔。对ROMK基因的检测发现,在四个巴特综合征家族中,与疾病共分离的突变会破坏ROMK功能。我们的研究结果确定了巴特综合征的遗传异质性,并证明了ROMK在体内的生理作用。

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