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青少年中枢性尿崩症的临床病程及病理生理研究:一例报告

The clinical course and pathophysiological investigation of adolescent gestational diabetes insipidus: a case report.

作者信息

Kondo Tatsuya, Nakamura Miwa, Kitano Sayaka, Kawashima Junji, Matsumura Takeshi, Ohba Takashi, Yamaguchi Munekage, Katabuchi Hidetaka, Araki Eiichi

机构信息

Department of Metabolic Medicine, Faculty of Life Sciences, Kumamoto University, 1-1-1 Chuo-Ward, Honjo, Kumamoto, 860-8556, Japan.

Department of Obstetrics and Gynecology, Faculty of Life Sciences, Kumamoto University, 1-1-1 Chuo-Ward, Honjo, Kumamoto, 860-8556, Japan.

出版信息

BMC Endocr Disord. 2018 Jan 30;18(1):4. doi: 10.1186/s12902-018-0234-6.

DOI:10.1186/s12902-018-0234-6
PMID:29378555
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC5789627/
Abstract

BACKGROUND

Gestational diabetes insipidus (GDI) is a rare endocrine complication during pregnancy that is associated with vasopressinase overproduction from the placenta. Although increased vasopressinase is associated with placental volume, the regulation of placental growth in the later stage of pregnancy is not well known.

CASE PRESENTATION

A 16-year-old pregnant woman was urgently transferred to our hospital because of threatened premature labor when the Kumamoto earthquakes hit the area where she lived. During her hospitalization, she complained of gradually increasing symptoms of polyuria and polydipsia. The serum level of arginine vasopressin (AVP) was 1.7 pg/mL, which is inconsistent with central DI. The challenge of diagnostic treatment using oral 1-deamino-8-D-AVP (DDAVP) successfully controlled her urine and allowed for normal delivery. DDAVP tablets were not necessary to control her polyuria thereafter. Based on these observations, clinical diagnosis of GDI was confirmed. Pathophysiological analyses revealed that vasopressinase expression was more abundant in the GDI patient's syncytiotrophoblast in placenta compared with that in a control subject. Serum vasopressinase was also observed during gestation and disappeared soon after delivery. Vasopressinase is reportedly identical to oxytocinase or insulin regulated aminopeptidase (IRAP), which is an abundant cargo protein associated with the glucose transporter 4 (GLUT4) storage vesicle. Interestingly, the expression and subcellular localization of GLUT4 appeared to occur in a vasopressinase (IRAP)-dependent manner.

CONCLUSION

Because placental volume may be associated with vasopressinase overproduction in GDI, vasopressinase (IRAP)/GLUT4 association appears to contribute to the growth of placenta in this case.

摘要

背景

妊娠性尿崩症(GDI)是孕期一种罕见的内分泌并发症,与胎盘产生过量的血管加压素酶有关。尽管血管加压素酶增加与胎盘体积有关,但妊娠后期胎盘生长的调节机制尚不清楚。

病例介绍

一名16岁孕妇在熊本地震袭击其居住地区时因先兆早产被紧急转至我院。住院期间,她抱怨多尿和烦渴症状逐渐加重。精氨酸血管加压素(AVP)血清水平为1.7 pg/mL,与中枢性尿崩症不符。口服1-去氨基-8-D-精氨酸加压素(DDAVP)进行诊断性治疗成功控制了她的尿量,并使其正常分娩。此后无需使用DDAVP片剂来控制她的多尿。基于这些观察结果,确诊为GDI。病理生理分析显示,与对照受试者相比,GDI患者胎盘合体滋养层中血管加压素酶表达更为丰富。妊娠期间也观察到血清血管加压素酶,分娩后不久消失。据报道,血管加压素酶与催产素酶或胰岛素调节氨肽酶(IRAP)相同,后者是一种与葡萄糖转运蛋白4(GLUT4)储存囊泡相关的丰富货物蛋白。有趣的是,GLUT4的表达和亚细胞定位似乎以血管加压素酶(IRAP)依赖的方式发生。

结论

由于胎盘体积可能与GDI中血管加压素酶产生过多有关,血管加压素酶(IRAP)/GLUT4关联在本病例中似乎有助于胎盘生长。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/26e3/5789627/166de29089c6/12902_2018_234_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/26e3/5789627/6bd1dda2046a/12902_2018_234_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/26e3/5789627/166de29089c6/12902_2018_234_Fig2_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/26e3/5789627/6bd1dda2046a/12902_2018_234_Fig1_HTML.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/26e3/5789627/166de29089c6/12902_2018_234_Fig2_HTML.jpg

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