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戈登综合征的基因型-表型相关性:两例携带新的杂合突变的病例报告。

Genotype-phenotype correlation in Gordon's syndrome: report of two cases carrying novel heterozygous mutations.

机构信息

Nephrology, Dialysis and Transplantation Unit, Department of Medicine-DIMED, University of Padua, Via Giustiniani n° 2, 35128, Padua, Italy.

Clinical Genetics Unit, Department of Women's and Children's Health, University of Padua, Padua, Italy.

出版信息

J Nephrol. 2022 Apr;35(3):859-862. doi: 10.1007/s40620-021-01083-1. Epub 2021 Jun 5.

Abstract

Gordon's syndrome, known also as Pseudohypoaldosteronism type II is a rare inherited dominant form of low-renin hypertension associated with hyperkalemia and metabolic acidosis. Four genes related to the regulation of the NaCl co-symporter NCC have been discovered associated to Gordon phenotypes: WINK 1 and WINK4, which, along with WNK2 and WNK3, encode a family of WNK-kinases, and KLHL3 and CUL3 encoding respectively, Kelch-like 3 protein and cullin. Heterozygous mutations in these genes constitutively activate NCC leading to abnormally increased salt reabsorption and salt-sensitive hypertension. Thiazide diuretic is the recognized treatment for this condition. We report and discuss phenotypic and genetic heterogeneity of two patients with Gordon's syndrome carrying novel heterozygous mutations in the WNK1 and KLHL3 genes. A very rare variant in the SCNN1G gene encoding the γ subunit of epithelial sodium channel ENaC was also identified in one patient.

摘要

高登综合征,亦称假性醛固酮减少症Ⅱ型,是一种罕见的遗传性显性低肾素型高血压,常伴有高钾血症和代谢性酸中毒。现已发现四个与 NaCl 协同转运蛋白 NCC 调节有关的基因与高登表型相关:WINK1 和 WINK4,与 WNK2 和 WNK3 一起,编码 WNK-激酶家族,以及 KLHL3 和 CUL3,分别编码 Kelch 样 3 蛋白和 cullin。这些基因的杂合突变持续激活 NCC,导致异常增加的盐重吸收和盐敏感性高血压。噻嗪类利尿剂是治疗这种疾病的公认方法。我们报告并讨论了两名携带 WNK1 和 KLHL3 基因突变的高登综合征患者的表型和遗传异质性。在一名患者中还发现了编码上皮钠通道 ENaC γ 亚单位的 SCNN1G 基因的一种非常罕见的变异。

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