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

• Published in: BMC endocrine disorders Vol. 18; no. 1; pp. 4 - 8
• Main Authors: Kondo, Tatsuya, Nakamura, Miwa, Kitano, Sayaka, Kawashima, Junji, Matsumura, Takeshi, Ohba, Takashi, Yamaguchi, Munekage, Katabuchi, Hidetaka, Araki, Eiichi
• Format: Journal Article
• Language: English
• Published: London BioMed Central 30.01.2018; BioMed Central Ltd; BMC
• Subjects: Basic and Clinical Endocrinology; Care and treatment; Case Report; Diabetes; Diabetes insipidus; Diagnosis; Endocrinology; Gestational diabetes; Gestational diabetes insipidus; Glucose transporter 4; Medicine; Medicine & Public Health; Metabolic Diseases; Risk factors; rology; Vasopressinase (=insulin regulated aminopeptidase: IRAP); Japan; Vasopressinase (=insulin regulated aminopeptidase: IRAP); Glucose transporter 4; Gestational diabetes insipidus
• ISSN: 1472-6823; 1472-6823
• DOI: 10.1186/s12902-018-0234-6

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.