Thakker R V
MRC Clinical Sciences Centre, Imperial College School of Medicine, Hammersmith Hospital, London, England.
Adv Nephrol Necker Hosp. 1999;29:289-98.
Recent studies of hereditary renal tubular disorders have facilitated the identification and roles of chloride channels and cotransporters in the regulation of the most abundant anion, Cl-, in the ECF. Thus, mutations that result in a loss of function of the voltage-gated chloride channel, CLC-5, are associated with Dent's disease, which is characterized by low-molecular weight proteinuria, hypercalciuria, nephrolithiasis, and renal failure. Mutations of another voltage-gated chloride channel, CLC-Kb, are associated with a form of Bartter's syndrome, whereas other forms of Bartter's syndrome are caused by mutations in the bumetanide-sensitive sodium-potassium-chloride cotransporter (NKCC2) and the potassium channel, ROMK. Finally, mutations of the thiazide-sensitive sodium-chloride cotransporter (NCCT) are associated with Gitelman's syndrome. These studies have helped to elucidate some of the renal tubular mechanisms regulating mineral homeostasis and the role of chloride channels.
最近对遗传性肾小管疾病的研究有助于确定氯离子通道和协同转运蛋白在调节细胞外液中最丰富的阴离子Cl-方面的作用。因此,导致电压门控氯离子通道CLC-5功能丧失的突变与丹特病相关,其特征为低分子量蛋白尿、高钙尿症、肾结石和肾衰竭。另一种电压门控氯离子通道CLC-Kb的突变与一种巴特综合征相关,而其他形式的巴特综合征则由布美他尼敏感的钠-钾-氯协同转运蛋白(NKCC2)和钾通道ROMK的突变引起。最后,噻嗪类敏感的钠-氯协同转运蛋白(NCCT)的突变与吉特曼综合征相关。这些研究有助于阐明一些调节矿物质稳态的肾小管机制以及氯离子通道的作用。