[AQP2新纯合突变p.R113C引起精氨酸抗利尿素耐药的临床和实验室特点]。

Q4 Medicine
N A Makretskaya, U S Nanzanova, I R Hamaganova, E R Eremina, A N Tiulpakov
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引用次数: 1

摘要

先天性肾源性尿崩症(CNDI,精氨酸抗利尿素)是一种罕见的遗传性疾病,其特征是肾脏对利尿素的抗利尿作用不敏感。NDI的临床特征为多尿伴少尿、夜尿和烦渴。在大多数病例中(约90%),肾源性尿崩症是一种由AVP V2受体基因(AVPR2)突变引起的x连锁隐性疾病。其余病例中,约10%为常染色体隐性遗传或显性遗传,据报道,这些患者的水通道蛋白2基因(AQP2)发生突变。迄今为止,在AQP2中登记的核苷酸变异是零星的,没有关于存在“频繁”突变和该疾病在全球人口和个别种族群体中流行的数据。本文报道了布里亚特共和国土著人群中由AQP2基因p.R113C纯合突变引起的12例精氨酸抗利尿素耐药病例。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

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

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

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.

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来源期刊
Problemy endokrinologii
Problemy endokrinologii Medicine-Endocrinology, Diabetes and Metabolism
CiteScore
1.40
自引率
0.00%
发文量
59
期刊介绍: Since 1955 the “Problems of Endocrinology” (or “Problemy Endocrinologii”) Journal publishes timely articles, balancing both clinical and experimental research, case reports, reviews and lectures on pressing problems of endocrinology. The Journal is aimed to the most topical issues of endocrinology: to chemical structure, biosynthesis and metabolism of hormones, the mechanism of their action at cellular and molecular level; pathogenesis and to clinic of the endocrine diseases, new methods of their diagnostics and treatment. The Journal: features original national and foreign research articles, reflecting world endocrinology development; issues thematic editions on specific areas; publishes chronicle of major international congress sessions and workshops on endocrinology, as well as state-of-the-art guidelines; is intended for scientists, endocrinologists diabetologists and specialists of allied trade, general practitioners, family physicians and pediatrics.
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