A case of central diabetes insipidus after COVID-19 as a probable diagnosis of lymphocytic infundibulo-neurohypophysitis with positive anti-rabphilin-3A antibodies with review of literature

Coronavirus disease 2019 (COVID-19) is caused by severe acute respiratory syndrome coronavirus 2, and various complications have been reported. Furthermore, there have been increasing reports of endocrinopathy related to COVID-19 following the pandemic. We report a 49-year-old healthy woman who deve...

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Main Authors: Yuka Natsuki, Yuki Nagata, Toshiki Nagasaki, Mari Morimoto, Norikazu Toi, Masafumi Kurajoh, Tomoaki Morioka, Tetsuo Shoji, Yasuo Imanishi, Naoko Iwata, Haruki Fujisawa, Atsushi Suzuki, Yoshihisa Sugimura, Masanori Emoto
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Language:English
Published: The Japan Endocrine Society 2024-12-01
Series:Endocrine Journal
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Online Access:https://www.jstage.jst.go.jp/article/endocrj/71/12/71_EJ24-0093/_html/-char/en
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author Yuka Natsuki
Yuki Nagata
Toshiki Nagasaki
Mari Morimoto
Norikazu Toi
Masafumi Kurajoh
Tomoaki Morioka
Tetsuo Shoji
Yasuo Imanishi
Naoko Iwata
Haruki Fujisawa
Atsushi Suzuki
Yoshihisa Sugimura
Masanori Emoto
author_facet Yuka Natsuki
Yuki Nagata
Toshiki Nagasaki
Mari Morimoto
Norikazu Toi
Masafumi Kurajoh
Tomoaki Morioka
Tetsuo Shoji
Yasuo Imanishi
Naoko Iwata
Haruki Fujisawa
Atsushi Suzuki
Yoshihisa Sugimura
Masanori Emoto
author_sort Yuka Natsuki
collection DOAJ
description Coronavirus disease 2019 (COVID-19) is caused by severe acute respiratory syndrome coronavirus 2, and various complications have been reported. Furthermore, there have been increasing reports of endocrinopathy related to COVID-19 following the pandemic. We report a 49-year-old healthy woman who developed rapid onset of polydipsia and polyuria three weeks after COVID-19. Laboratory tests indicated low urine osmolarity and increased serum osmolarity, and antidiuretic hormone (ADH) was undetectable. Urine osmolality remained low with water deprivation. Similarly, plasma ADH responses to hypertonic-saline infusion were blunted and urine osmolality increased in response to desmopressin. There was no clear evidence of anterior pituitary dysfunction. T1-weighted magnetic resonance imaging (MRI) showed pituitary stalk thickening and absence of posterior pituitary bright signal spots, suggesting the presence of hypophysitis. Based on these results, we made a probable diagnosis of lymphocytic infundibulo-neurohypophysitis (LINH) which have caused central diabetes insipidus. Positive findings for serum anti-rabphilin-3A antibodies, reported as a potential diagnostic marker for LINH, were also noted. Following oral desmopressin administration, polydipsia and polyuria were quickly improved, though treatment with desmopressin was still required over four months. This is the first report of a patient with a probable diagnosis of LINH after COVID-19 who tested positive for anti-rabphilin-3A antibodies. Positive findings for those antibodies suggest that pituitary dysfunction associated with COVID-19 is hypophysitis involving an abnormal immune mechanism. The presence of anti-rabphilin-3A antibodies may be useful as a non-invasive diagnostic marker of LINH and potentially serve as a valuable diagnostic aid in cases of LINH associated with COVID-19.
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series Endocrine Journal
spelling doaj-art-a2ba444135664653839d77f315f19fde2025-01-22T05:34:07ZengThe Japan Endocrine SocietyEndocrine Journal1348-45402024-12-0171121165117310.1507/endocrj.EJ24-0093endocrjA case of central diabetes insipidus after COVID-19 as a probable diagnosis of lymphocytic infundibulo-neurohypophysitis with positive anti-rabphilin-3A antibodies with review of literatureYuka Natsuki0Yuki Nagata1Toshiki Nagasaki2Mari Morimoto3Norikazu Toi4Masafumi Kurajoh5Tomoaki Morioka6Tetsuo Shoji7Yasuo Imanishi8Naoko Iwata9Haruki Fujisawa10Atsushi Suzuki11Yoshihisa Sugimura12Masanori Emoto13Department of Metabolism, Endocrinology and Molecular Medicine, Osaka Metropolitan University Graduate School of Medicine, Osaka 545-8585, JapanDepartment of Vascular Medicine, Osaka Metropolitan University Graduate School of Medicine, Osaka 545-8585, JapanNagasaki Thyroid Clinic, Osaka 546-0014, JapanDepartment of Metabolism, Endocrinology and Molecular Medicine, Osaka Metropolitan University Graduate School of Medicine, Osaka 545-8585, JapanDepartment of Metabolism, Endocrinology and Molecular Medicine, Osaka Metropolitan University Graduate School of Medicine, Osaka 545-8585, JapanDepartment of Metabolism, Endocrinology and Molecular Medicine, Osaka Metropolitan University Graduate School of Medicine, Osaka 545-8585, JapanDepartment of Metabolism, Endocrinology and Molecular Medicine, Osaka Metropolitan University Graduate School of Medicine, Osaka 545-8585, JapanDepartment of Vascular Medicine, Osaka Metropolitan University Graduate School of Medicine, Osaka 545-8585, JapanDepartment of Metabolism, Endocrinology and Molecular Medicine, Osaka Metropolitan University Graduate School of Medicine, Osaka 545-8585, JapanDepartment of Endocrinology, Diabetes and Metabolism, Fujita Health University School of Medicine, Aichi 470-1192, JapanDepartment of Endocrinology, Diabetes and Metabolism, Fujita Health University School of Medicine, Aichi 470-1192, JapanDepartment of Endocrinology, Diabetes and Metabolism, Fujita Health University School of Medicine, Aichi 470-1192, JapanDepartment of Endocrinology, Diabetes and Metabolism, Fujita Health University School of Medicine, Aichi 470-1192, JapanDepartment of Metabolism, Endocrinology and Molecular Medicine, Osaka Metropolitan University Graduate School of Medicine, Osaka 545-8585, JapanCoronavirus disease 2019 (COVID-19) is caused by severe acute respiratory syndrome coronavirus 2, and various complications have been reported. Furthermore, there have been increasing reports of endocrinopathy related to COVID-19 following the pandemic. We report a 49-year-old healthy woman who developed rapid onset of polydipsia and polyuria three weeks after COVID-19. Laboratory tests indicated low urine osmolarity and increased serum osmolarity, and antidiuretic hormone (ADH) was undetectable. Urine osmolality remained low with water deprivation. Similarly, plasma ADH responses to hypertonic-saline infusion were blunted and urine osmolality increased in response to desmopressin. There was no clear evidence of anterior pituitary dysfunction. T1-weighted magnetic resonance imaging (MRI) showed pituitary stalk thickening and absence of posterior pituitary bright signal spots, suggesting the presence of hypophysitis. Based on these results, we made a probable diagnosis of lymphocytic infundibulo-neurohypophysitis (LINH) which have caused central diabetes insipidus. Positive findings for serum anti-rabphilin-3A antibodies, reported as a potential diagnostic marker for LINH, were also noted. Following oral desmopressin administration, polydipsia and polyuria were quickly improved, though treatment with desmopressin was still required over four months. This is the first report of a patient with a probable diagnosis of LINH after COVID-19 who tested positive for anti-rabphilin-3A antibodies. Positive findings for those antibodies suggest that pituitary dysfunction associated with COVID-19 is hypophysitis involving an abnormal immune mechanism. The presence of anti-rabphilin-3A antibodies may be useful as a non-invasive diagnostic marker of LINH and potentially serve as a valuable diagnostic aid in cases of LINH associated with COVID-19.https://www.jstage.jst.go.jp/article/endocrj/71/12/71_EJ24-0093/_html/-char/encoronavirus disease 2019 (covid-19)lymphocytic infundibulo-neurohypophysitiscentral diabetes insipidusanti-rabphilin-3a antibodies
spellingShingle Yuka Natsuki
Yuki Nagata
Toshiki Nagasaki
Mari Morimoto
Norikazu Toi
Masafumi Kurajoh
Tomoaki Morioka
Tetsuo Shoji
Yasuo Imanishi
Naoko Iwata
Haruki Fujisawa
Atsushi Suzuki
Yoshihisa Sugimura
Masanori Emoto
A case of central diabetes insipidus after COVID-19 as a probable diagnosis of lymphocytic infundibulo-neurohypophysitis with positive anti-rabphilin-3A antibodies with review of literature
Endocrine Journal
coronavirus disease 2019 (covid-19)
lymphocytic infundibulo-neurohypophysitis
central diabetes insipidus
anti-rabphilin-3a antibodies
title A case of central diabetes insipidus after COVID-19 as a probable diagnosis of lymphocytic infundibulo-neurohypophysitis with positive anti-rabphilin-3A antibodies with review of literature
title_full A case of central diabetes insipidus after COVID-19 as a probable diagnosis of lymphocytic infundibulo-neurohypophysitis with positive anti-rabphilin-3A antibodies with review of literature
title_fullStr A case of central diabetes insipidus after COVID-19 as a probable diagnosis of lymphocytic infundibulo-neurohypophysitis with positive anti-rabphilin-3A antibodies with review of literature
title_full_unstemmed A case of central diabetes insipidus after COVID-19 as a probable diagnosis of lymphocytic infundibulo-neurohypophysitis with positive anti-rabphilin-3A antibodies with review of literature
title_short A case of central diabetes insipidus after COVID-19 as a probable diagnosis of lymphocytic infundibulo-neurohypophysitis with positive anti-rabphilin-3A antibodies with review of literature
title_sort case of central diabetes insipidus after covid 19 as a probable diagnosis of lymphocytic infundibulo neurohypophysitis with positive anti rabphilin 3a antibodies with review of literature
topic coronavirus disease 2019 (covid-19)
lymphocytic infundibulo-neurohypophysitis
central diabetes insipidus
anti-rabphilin-3a antibodies
url https://www.jstage.jst.go.jp/article/endocrj/71/12/71_EJ24-0093/_html/-char/en
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