クボクラ（イシカワ）　マユミ   Ishikawa-Kubokura Mayumi   窪倉（石川）　真由美    所属   東邦大学  医学部 医学科（大森病院）    職種   教授
論文種別	原著
言語種別	英語
査読の有無	査読なし
表題	A Case of Adrenocorticotropin-dependent Cushing Syndrome with Osilodrostat Exposure in Early Pregnancy.
掲載誌名	正式名：JCEM case reports
掲載区分	国外
巻・号・頁	3(12),pp.luaf269
著者・共著者	Yukako Nakayama,Mayumi Ishikawa,Asako Watanabe,Yoshio Shima,Shigeyuki Tahara,Masato Iwabu
発行年月	2025/12
概要	Osilodrostat is a novel treatment for adrenocorticotropin-dependent Cushing syndrome; however, its safety during pregnancy has not been reported. This case involves a patient with Cushing disease who became pregnant while on osilodrostat. She was diagnosed at 31 years of age and underwent pituitary tumor removal. After a relapse at 35 years of age, she was initially treated with metyrapone but switched to osilodrostat and hydrocortisone because of nausea, achieving reasonable cortisol control. At 37 years of age, she unknowingly became pregnant despite irregular periods, and the pregnancy was detected at 16 weeks because of ongoing nausea. Osilodrostat was stopped, and she was started on pasireotide and metyrapone. The pregnancy proceeded normally despite elevated urinary free cortisol levels, although she contracted COVID-19 at 25 weeks. At 26 weeks and 1 day, preterm rupture of membranes and breech presentation led to an emergency cesarean section. The newborn had no adrenal insufficiency and developed normally. This case prompts consideration of whether osilodrostat can be used during pregnancy if risks are justified. Pasireotide is rarely used in pregnancy and may have limited effectiveness, but when given, can cause hyperglycemia because of insulin and incretin suppression and should be monitored carefully.
DOI	10.1210/jcemcr/luaf269
PMID	41267803