Klepalova V V, Pushkareva O S, Iziurova N V, Aksenov A V
South Ural State Medical University.
Probl Endokrinol (Mosk). 2022 Jul 13;68(5):87-90. doi: 10.14341/probl13098.
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.
尿崩症的患病率呈全球上升趋势。X连锁遗传的肾性尿崩症症状出现在男性以及具有杂合突变的女性中,其特征为多尿、烦渴、低比重尿这一孤立的症状群。与成人相比,儿童在限液时更常出现缺水性脱水的临床表现,伴有高钠血症、高热和血浆渗透压升高。本文报告一例男性患者的X连锁家族性肾性尿崩症病例。同时,在母系家族中,母亲和祖母也有饮水需求增加的情况,使用去氨加压素无效。哥哥和弟弟自婴儿期起就出现了以严重口渴和多尿为表现形式的尿崩症临床表现,经检查后确诊为尿崩症并开具了去氨加压素。由于使用去氨加压素无效,通过聚合酶链反应(PCR)及随后的直接测序对水通道蛋白2(AQP2)基因和血管加压素2型受体(AVPR2)基因的外显子及内含子相邻区域进行了分析。在AQP2基因中未发现突变。在AVPR2基因中发现了半合子替代S315I。确诊为X连锁家族性肾性尿崩症。建议使用氢氯噻嗪,在持续服用该药的背景下仅观察到轻微的积极趋势。