A Pregnant Woman with Excess Vasopressinase-Induced Diabetes Insipidus Complicated by Central Diabetes Insipidus like Lymphocytic Infundibulo-Neurohypophysitis
Background. Gestational diabetes insipidus (DI) is a very rare complication of pregnancy. We present a case of gestational DI combining two different types of DI. Case Presentation. A 39-year-old pregnant woman suddenly presented with thirst, polydipsia, and polyuria after 31 gestation weeks (GWs)....
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| Format: | Article |
| Language: | English |
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Wiley
2024-01-01
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| Series: | Case Reports in Endocrinology |
| Online Access: | http://dx.doi.org/10.1155/2024/8687054 |
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| author | Shinnosuke Yanagisawa Yoichi Oikawa Mai Endo Kazuyuki Inoue Ritsuko Nakajima Shigemitsu Yasuda Masayasu Sato Naoko Iwata Haruki Fujisawa Atsushi Suzuki Yoshihisa Sugimura Masashi Isshiki Akira Shimada |
| author_facet | Shinnosuke Yanagisawa Yoichi Oikawa Mai Endo Kazuyuki Inoue Ritsuko Nakajima Shigemitsu Yasuda Masayasu Sato Naoko Iwata Haruki Fujisawa Atsushi Suzuki Yoshihisa Sugimura Masashi Isshiki Akira Shimada |
| author_sort | Shinnosuke Yanagisawa |
| collection | DOAJ |
| description | Background. Gestational diabetes insipidus (DI) is a very rare complication of pregnancy. We present a case of gestational DI combining two different types of DI. Case Presentation. A 39-year-old pregnant woman suddenly presented with thirst, polydipsia, and polyuria after 31 gestation weeks (GWs). Based on laboratory findings of hypotonic urine (78 mOsm/kgH2O) with higher plasma osmolality (298 mOsm/kgH2O) and higher serum sodium levels (149 mEq/L), gestational DI was suspected, and the clinical course was monitored without therapy until the results of a measurement of plasma arginine vasopressin (AVP) levels were available. However, she subsequently developed acute prerenal failure and underwent an emergency cesarean section at 34 GWs. Her resected placenta weighed 920 g, nearly twice the normal weight. Immediately following delivery, intranasal 1-desamino-8-D-arginine vasopressin was administered, and her symptoms promptly disappeared. Afterward, her predelivery plasma AVP level was found to have been inappropriately low (0.7 pg/mL) given her serum sodium level. The patient’s serum vasopressinase level just before delivery was 2,855 ng/mL, more than 1,000 times the upper limit of the normal range, suggesting excess vasopressinase-induced DI. The presence of anti-rabphilin-3A antibodies in the patient’s blood, a hypertonic saline infusion test result, and loss of the high-intensity signal of the posterior pituitary on fat-suppressed T1-weighted magnetic resonance images without thickening of the stalk and enlargement of the neurohypophysis suggested concurrent central DI-like lymphocytic infundibulo-neurohypophysitis (LINH). Conclusion. In addition to the degradation of AVP by excess placental vasopressinase due to the enlarged placenta, an insufficient compensatory increase in AVP secretion from the posterior pituitary gland due to LINH-like pathogenesis might have led to DI symptoms. |
| format | Article |
| id | doaj-art-21691468379c48a5afc60bdbfea8e542 |
| institution | Kabale University |
| issn | 2090-651X |
| language | English |
| publishDate | 2024-01-01 |
| publisher | Wiley |
| record_format | Article |
| series | Case Reports in Endocrinology |
| spelling | doaj-art-21691468379c48a5afc60bdbfea8e5422025-02-03T05:54:44ZengWileyCase Reports in Endocrinology2090-651X2024-01-01202410.1155/2024/8687054A Pregnant Woman with Excess Vasopressinase-Induced Diabetes Insipidus Complicated by Central Diabetes Insipidus like Lymphocytic Infundibulo-NeurohypophysitisShinnosuke Yanagisawa0Yoichi Oikawa1Mai Endo2Kazuyuki Inoue3Ritsuko Nakajima4Shigemitsu Yasuda5Masayasu Sato6Naoko Iwata7Haruki Fujisawa8Atsushi Suzuki9Yoshihisa Sugimura10Masashi Isshiki11Akira Shimada12Department of Endocrinology and DiabetesDepartment of Endocrinology and DiabetesDepartment of Endocrinology and DiabetesDepartment of Endocrinology and DiabetesDepartment of Endocrinology and DiabetesDepartment of Endocrinology and DiabetesDepartment of Obstetrics and GynecologyDepartment of Endocrinology and DiabetesDepartment of EndocrinologyDepartment of EndocrinologyDepartment of EndocrinologyDepartment of Endocrinology and DiabetesDepartment of Endocrinology and DiabetesBackground. Gestational diabetes insipidus (DI) is a very rare complication of pregnancy. We present a case of gestational DI combining two different types of DI. Case Presentation. A 39-year-old pregnant woman suddenly presented with thirst, polydipsia, and polyuria after 31 gestation weeks (GWs). Based on laboratory findings of hypotonic urine (78 mOsm/kgH2O) with higher plasma osmolality (298 mOsm/kgH2O) and higher serum sodium levels (149 mEq/L), gestational DI was suspected, and the clinical course was monitored without therapy until the results of a measurement of plasma arginine vasopressin (AVP) levels were available. However, she subsequently developed acute prerenal failure and underwent an emergency cesarean section at 34 GWs. Her resected placenta weighed 920 g, nearly twice the normal weight. Immediately following delivery, intranasal 1-desamino-8-D-arginine vasopressin was administered, and her symptoms promptly disappeared. Afterward, her predelivery plasma AVP level was found to have been inappropriately low (0.7 pg/mL) given her serum sodium level. The patient’s serum vasopressinase level just before delivery was 2,855 ng/mL, more than 1,000 times the upper limit of the normal range, suggesting excess vasopressinase-induced DI. The presence of anti-rabphilin-3A antibodies in the patient’s blood, a hypertonic saline infusion test result, and loss of the high-intensity signal of the posterior pituitary on fat-suppressed T1-weighted magnetic resonance images without thickening of the stalk and enlargement of the neurohypophysis suggested concurrent central DI-like lymphocytic infundibulo-neurohypophysitis (LINH). Conclusion. In addition to the degradation of AVP by excess placental vasopressinase due to the enlarged placenta, an insufficient compensatory increase in AVP secretion from the posterior pituitary gland due to LINH-like pathogenesis might have led to DI symptoms.http://dx.doi.org/10.1155/2024/8687054 |
| spellingShingle | Shinnosuke Yanagisawa Yoichi Oikawa Mai Endo Kazuyuki Inoue Ritsuko Nakajima Shigemitsu Yasuda Masayasu Sato Naoko Iwata Haruki Fujisawa Atsushi Suzuki Yoshihisa Sugimura Masashi Isshiki Akira Shimada A Pregnant Woman with Excess Vasopressinase-Induced Diabetes Insipidus Complicated by Central Diabetes Insipidus like Lymphocytic Infundibulo-Neurohypophysitis Case Reports in Endocrinology |
| title | A Pregnant Woman with Excess Vasopressinase-Induced Diabetes Insipidus Complicated by Central Diabetes Insipidus like Lymphocytic Infundibulo-Neurohypophysitis |
| title_full | A Pregnant Woman with Excess Vasopressinase-Induced Diabetes Insipidus Complicated by Central Diabetes Insipidus like Lymphocytic Infundibulo-Neurohypophysitis |
| title_fullStr | A Pregnant Woman with Excess Vasopressinase-Induced Diabetes Insipidus Complicated by Central Diabetes Insipidus like Lymphocytic Infundibulo-Neurohypophysitis |
| title_full_unstemmed | A Pregnant Woman with Excess Vasopressinase-Induced Diabetes Insipidus Complicated by Central Diabetes Insipidus like Lymphocytic Infundibulo-Neurohypophysitis |
| title_short | A Pregnant Woman with Excess Vasopressinase-Induced Diabetes Insipidus Complicated by Central Diabetes Insipidus like Lymphocytic Infundibulo-Neurohypophysitis |
| title_sort | pregnant woman with excess vasopressinase induced diabetes insipidus complicated by central diabetes insipidus like lymphocytic infundibulo neurohypophysitis |
| url | http://dx.doi.org/10.1155/2024/8687054 |
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