[肾源性x连锁糖尿病的家族形式]。

Q4 Medicine Problemy endokrinologii Pub Date : 2022-07-13 DOI:10.14341/probl13098
V V Klepalova, O S Pushkareva, N V Iziurova, A V Aksenov
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引用次数: 1

摘要

尿崩症的患病率呈全球上升趋势。具有x连锁遗传的肾源性尿崩症的症状出现在男性中,在具有杂合突变的女性中,其特征是多尿、多饮、尿后不足的孤立症状复合物。在儿童中,与成人相比,更常见的是液体限制,缺水性脱水的临床发展为高钠血症、高热和血浆高渗透压。这个手稿提出了一个病例肾源性尿崩症,x连锁家族形式的男性患者。与此同时,在女性系的家庭中,母亲和祖母对水的需求也增加了,使用minirin是无效的。哥哥和弟弟从幼年就注意到尿崩症的临床表现,表现为严重口渴和多尿,经检查,诊断为尿崩症,并开去氨加压素。由于去氨加压素的使用效果不明显,我们对AQP2和AVPR2基因的外显子和内含子邻近片段进行PCR分析,然后直接测序。AQP2基因未发现突变。在AVPR2基因中发现了半合子替换S315I。家族型X被证实与肾源性尿崩症相关。建议服用一种低噻嗪类药物,在持续摄入的背景下,只观察到轻微的阳性趋势。
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[Family form of nephrogenic X-linked diabetes indiabetes].

There is a global trend towards an increase in the prevalence of diabetes insipidus. Symptoms of nephrogenic diabetes insipidus with X-linked inheritance appear in men, in women with heterozygous mutations, are characterized by an isolated symptom complex of polyuria, polydipsia, hypostenuria. In children, more often than in adults, with fluid restriction, a clinic of water-deficient dehydration develops with hypernatremia, hyperthermia, and plasma hyperosmolality. This manuscript presents a case of Nephrogenic diabetes insipidus, X-linked familial form in male patients.At the same time, in the family along the female line, the mother and grandmother also had an increased need for water, the use of minirin was ineffective. In the older brother and younger brother, clinical manifestations of diabetes insipidus in the form of severe thirst and polyuria were noted from infancy, after the examination, the diagnosis was made - diabetes insipidus and desmopressin was prescribed.Due to the lack of effect from the use of desmopressin, the analysis of exons and adjacent sections of the introns of the AQP2 and AVPR2 genes was carried out by PCR and subsequent direct sequencing. No mutations were found in the AQP2 gene. The hemizygous substitution S315I was found in the AVPR2 gene. The familial form X was confirmed - linked nephrogenic diabetes insipidus. A hypothiazide was recommended, against the background of constant intake of which only a slight positive trend is observed.

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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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