Cushing’s disease with twin pregnancy and diabetes mellitus: a case report and literature review

A 38-year-old Japanese woman with a history of abnormal thyroid function of non-autoimmune origin, pituitary endocrine tumor, and untreated diabetes mellitus was referred to our outpatient clinic when she became pregnant with twins. Physical findings consistent with Cushing’s syndrome (CS) were abse...

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Main Authors: Hideyasu Asai, Ikuo Yamamori, Shigeru Hagimoto, Kyoichi Okumura, Koki Sakakibara
Format: Article
Language:English
Published: The Japan Endocrine Society 2024-10-01
Series:Endocrine Journal
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Online Access:https://www.jstage.jst.go.jp/article/endocrj/71/10/71_EJ23-0725/_html/-char/en
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author Hideyasu Asai
Ikuo Yamamori
Shigeru Hagimoto
Kyoichi Okumura
Koki Sakakibara
author_facet Hideyasu Asai
Ikuo Yamamori
Shigeru Hagimoto
Kyoichi Okumura
Koki Sakakibara
author_sort Hideyasu Asai
collection DOAJ
description A 38-year-old Japanese woman with a history of abnormal thyroid function of non-autoimmune origin, pituitary endocrine tumor, and untreated diabetes mellitus was referred to our outpatient clinic when she became pregnant with twins. Physical findings consistent with Cushing’s syndrome (CS) were absent at the time of presentation. Although baseline plasma adrenocorticotropic hormone, serum cortisol, and 24-hour urinary free cortisol excretion levels were above the upper limits of normal non-pregnant reference ranges, we could not exclude a physiological increase associated with pregnancy. No medical or surgical intervention for hypercortisolism was performed during pregnancy. Spontaneous vaginal delivery resulted in the normal delivery of live twins. A diagnosis of Cushing’s disease (CD) was established when papery skin developed postpartum. Transsphenoidal surgery was performed and the hypercortisolism partially resolved post-operatively. The patient’s abnormal thyroid function also resolved. Pregnancy in women with endogenous CS is rare, with less than 300 cases reported. Most reported cases of CS during pregnancy are of adrenal origin. Only two cases of twin pregnancies with CD have been reported. Therefore, we reported the third case of CD in a twin pregnancy and reviewed the diagnostic and therapeutic challenges associated with CD during pregnancy.
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institution Kabale University
issn 1348-4540
language English
publishDate 2024-10-01
publisher The Japan Endocrine Society
record_format Article
series Endocrine Journal
spelling doaj-art-0a406d7d575d42f0b372e88a0b44afb12025-01-22T05:22:57ZengThe Japan Endocrine SocietyEndocrine Journal1348-45402024-10-0171101003101110.1507/endocrj.EJ23-0725endocrjCushing’s disease with twin pregnancy and diabetes mellitus: a case report and literature reviewHideyasu Asai0Ikuo Yamamori1Shigeru Hagimoto2Kyoichi Okumura3Koki Sakakibara4Department of Endocrinology and Diabetes, Toyohashi Municipal Hospital, Aichi 441-8570, JapanDepartment of Endocrinology and Diabetes, Toyohashi Municipal Hospital, Aichi 441-8570, JapanDepartment of Endocrinology and Diabetes, Toyohashi Municipal Hospital, Aichi 441-8570, JapanDepartment of Endocrinology and Diabetes, Toyohashi Municipal Hospital, Aichi 441-8570, JapanDepartment of Endocrinology and Diabetes, Toyohashi Municipal Hospital, Aichi 441-8570, JapanA 38-year-old Japanese woman with a history of abnormal thyroid function of non-autoimmune origin, pituitary endocrine tumor, and untreated diabetes mellitus was referred to our outpatient clinic when she became pregnant with twins. Physical findings consistent with Cushing’s syndrome (CS) were absent at the time of presentation. Although baseline plasma adrenocorticotropic hormone, serum cortisol, and 24-hour urinary free cortisol excretion levels were above the upper limits of normal non-pregnant reference ranges, we could not exclude a physiological increase associated with pregnancy. No medical or surgical intervention for hypercortisolism was performed during pregnancy. Spontaneous vaginal delivery resulted in the normal delivery of live twins. A diagnosis of Cushing’s disease (CD) was established when papery skin developed postpartum. Transsphenoidal surgery was performed and the hypercortisolism partially resolved post-operatively. The patient’s abnormal thyroid function also resolved. Pregnancy in women with endogenous CS is rare, with less than 300 cases reported. Most reported cases of CS during pregnancy are of adrenal origin. Only two cases of twin pregnancies with CD have been reported. Therefore, we reported the third case of CD in a twin pregnancy and reviewed the diagnostic and therapeutic challenges associated with CD during pregnancy.https://www.jstage.jst.go.jp/article/endocrj/71/10/71_EJ23-0725/_html/-char/encushing’s diseasetwin pregnancydiabetes mellitus
spellingShingle Hideyasu Asai
Ikuo Yamamori
Shigeru Hagimoto
Kyoichi Okumura
Koki Sakakibara
Cushing’s disease with twin pregnancy and diabetes mellitus: a case report and literature review
Endocrine Journal
cushing’s disease
twin pregnancy
diabetes mellitus
title Cushing’s disease with twin pregnancy and diabetes mellitus: a case report and literature review
title_full Cushing’s disease with twin pregnancy and diabetes mellitus: a case report and literature review
title_fullStr Cushing’s disease with twin pregnancy and diabetes mellitus: a case report and literature review
title_full_unstemmed Cushing’s disease with twin pregnancy and diabetes mellitus: a case report and literature review
title_short Cushing’s disease with twin pregnancy and diabetes mellitus: a case report and literature review
title_sort cushing s disease with twin pregnancy and diabetes mellitus a case report and literature review
topic cushing’s disease
twin pregnancy
diabetes mellitus
url https://www.jstage.jst.go.jp/article/endocrj/71/10/71_EJ23-0725/_html/-char/en
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AT ikuoyamamori cushingsdiseasewithtwinpregnancyanddiabetesmellitusacasereportandliteraturereview
AT shigeruhagimoto cushingsdiseasewithtwinpregnancyanddiabetesmellitusacasereportandliteraturereview
AT kyoichiokumura cushingsdiseasewithtwinpregnancyanddiabetesmellitusacasereportandliteraturereview
AT kokisakakibara cushingsdiseasewithtwinpregnancyanddiabetesmellitusacasereportandliteraturereview