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巴特综合征和吉特曼综合征的病理生理及分子基础。

The pathophysiological and molecular basis of Bartter's and Gitelman's syndromes.

作者信息

Bhandari S

机构信息

School of Biomedical Sciences, Department of Physiology, University of Leeds, UK.

出版信息

Postgrad Med J. 1999 Jul;75(885):391-6. doi: 10.1136/pgmj.75.885.391.

Abstract

Molecular defects affecting the transport of sodium, potassium and chloride in the nephron through the ROMK K+ channel, Na+/K+/2Cl- cotransporter, the Na+/Cl- cotransporter and chloride channel have been identified in patients with Bartter's and Gitelman's syndromes. Defects of the angiotensin II type I receptor and CFTR have also being described. These defects are simple (i.e., most are single amino acid substitutions) but affect key elements in tubular transport. The simplicity of the genetic defects may explain why the inheritance of these conditions remains unclear in most kindreds (i.e., not just recessive or dominant) and emphasises the crucial importance of the conformational structure of these channels. Application of this molecular information will allow the early genetic identification of patients with these syndromes and enable us to differentiate between the various disorders at a functional level. It may also identify a subgroup in which the heterozygous form may make patients potentially exquisitely sensitive to diuretics.

摘要

在巴特综合征和吉特曼综合征患者中,已发现影响肾单位中钠、钾和氯通过ROMK钾通道、钠/钾/2氯协同转运蛋白、钠/氯协同转运蛋白和氯通道进行转运的分子缺陷。也有关于血管紧张素II 1型受体和囊性纤维化跨膜传导调节因子缺陷的描述。这些缺陷很简单(即大多数是单个氨基酸替换),但会影响肾小管转运的关键元件。遗传缺陷的简单性可能解释了为什么在大多数家族中这些病症的遗传方式仍不明确(即不只是隐性或显性),并强调了这些通道构象结构的至关重要性。应用这些分子信息将有助于对这些综合征患者进行早期基因鉴定,并使我们能够在功能水平上区分各种疾病。它还可能识别出一个亚组,其中杂合形式可能使患者对利尿剂极度敏感。

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