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[由水通道蛋白2(AQP2)中新的纯合突变p.R113C引起的精氨酸加压素抵抗的临床和实验室特征]

[Clinical and laboratory characteristics of arginine vasopressin resistance, caused by a new homozygous mutation p.R113C in AQP2].

作者信息

Makretskaya N A, Nanzanova U S, Hamaganova I R, Eremina E R, Tiulpakov A N

机构信息

Research Centre for Medical Genetics.

Children's Republican Clinical Hospital.

出版信息

Probl Endokrinol (Mosk). 2023 May 12;69(2):75-79. doi: 10.14341/probl13188.

Abstract

Congenital nephrogenic diabetes insipidus (CNDI, arginine vasopressin resistance) is a rare inherited disorder characterized by insensitivity of the kidney to the antidiuretic effect of vasopressin. NDI is clinically characterized by polyuria with hyposthenuria and nocturia and polydipsia. In the majority of cases, about 90%, nephrogenic diabetes insipidus is an X-linked recessive disorder caused by mutations in the AVP V2 receptor gene (AVPR2). In the remaining cases, about 10%, the disease is autosomal recessive or dominant and, for these patients, mutations in the aquaporin 2 gene (AQP2) have been reported. To date, the nucleotide variants registered in AQP2 were sporadic, there is no data on the presence of «frequent» mutations and the prevalence of the disease both among the global population and among individual ethnic groups. In this paper, we describe 12 cases of arginine vasopressin resistance caused by a new homozygous mutation p.R113C in AQP2 presented among the indigenous population of the Republic of Buryatia.

摘要

先天性肾性尿崩症(CNDI,精氨酸加压素抵抗)是一种罕见的遗传性疾病,其特征是肾脏对加压素的抗利尿作用不敏感。肾性尿崩症的临床特征为多尿伴低渗尿、夜尿症和烦渴。在大多数病例中,约90%,肾性尿崩症是一种X连锁隐性疾病,由精氨酸加压素V2受体基因(AVPR2)突变引起。在其余约10%的病例中,该疾病为常染色体隐性或显性,对于这些患者,已报道水通道蛋白2基因(AQP2)存在突变。迄今为止,AQP2中登记的核苷酸变异是散发性的,没有关于“常见”突变的存在以及该疾病在全球人群和各个种族群体中的患病率的数据。在本文中,我们描述了12例由AQP2中一种新的纯合突变p.R113C引起的精氨酸加压素抵抗病例,这些病例出现在布里亚特共和国的原住民中。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/efaf/10204789/cabd555d1e00/problendo-69-13188-g001.jpg

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