{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,2]],"date-time":"2026-06-02T15:19:55Z","timestamp":1780413595550,"version":"3.54.1"},"reference-count":17,"publisher":"The Endocrine Society","issue":"4","license":[{"start":{"date-parts":[[2025,3,20]],"date-time":"2025-03-20T00:00:00Z","timestamp":1742428800000},"content-version":"vor","delay-in-days":3,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"name":"National Cancer Institute Cancer Center","award":["P30 CA008748"],"award-info":[{"award-number":["P30 CA008748"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,3,17]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Cushing syndrome (CS) in pregnancy is a rare disorder with challenging diagnostic and therapeutic considerations. For patients with medically managed CS planning pregnancy, bilateral adrenalectomy is advised before conception. To our knowledge, no case of Cushing disease (CD) medically treated throughout pregnancy with cortisol synthesis inhibition has been described. We present 2 cases of CD in pregnancy managed with metyrapone, a non-Food and Drug Administration-approved drug for pregnancy. The first case involves a 31-year-old woman with recurrent CD who conceived twins via in vitro fertilization while on metyrapone. She had an uneventful pregnancy, delivering healthy twins at 37 weeks. Postpartum, she developed hypertension, which resolved spontaneously, and remained on metyrapone, with stable cortisol levels and normal development of her twins. The second case describes a 29-year-old woman with recurrent CD since adolescence, status post 2 transsphenoidal resections and gamma knife radiosurgery, who conceived naturally on metyrapone. After an uncomplicated pregnancy, she developed postpartum preeclampsia requiring temporary antihypertensive therapy. Both cases showed favorable maternal and fetal outcomes, highlighting metyrapone as a therapeutic option for managing CD during pregnancy.<\/jats:p>","DOI":"10.1210\/jcemcr\/luaf042","type":"journal-article","created":{"date-parts":[[2025,3,20]],"date-time":"2025-03-20T23:57:17Z","timestamp":1742515037000},"source":"Crossref","is-referenced-by-count":2,"title":["Successful Medical Management of Cushing Disease With Metyrapone Throughout Pregnancy: A Report of 2 Cases"],"prefix":"10.1210","volume":"3","author":[{"given":"Angeliki","family":"Theodorou","sequence":"first","affiliation":[{"name":"Division of Endocrinology, Department of Medicine, Memorial Sloan Kettering Cancer Center , New York, NY 10021 ,","place":["USA"]},{"name":"Department of Radiology, Columbia University Irving Medical Center , New York, NY 10032 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3722-1889","authenticated-orcid":false,"given":"Eliza B","family":"Geer","sequence":"additional","affiliation":[{"name":"Department of Medicine and Neurosurgery, Multidisciplinary Pituitary and Skull Base Tumor Center, Memorial Sloan Kettering Cancer Center , New York, NY 10021 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"80","published-online":{"date-parts":[[2025,3,20]]},"reference":[{"issue":"3","key":"2025032012384026300_luaf042-B1","doi-asserted-by":"crossref","first-page":"188","DOI":"10.1016\/j.ando.2022.04.001","article-title":"Impact of Cushing's syndrome on fertility and pregnancy","volume":"83","author":"Castinetti","year":"2022","journal-title":"Ann Endocrinol (Paris)"},{"issue":"2","key":"2025032012384026300_luaf042-B2","doi-asserted-by":"crossref","first-page":"555","DOI":"10.1007\/s12020-016-1117-0","article-title":"Cushing's syndrome and pregnancy outcomes: a systematic review of published cases","volume":"55","author":"Caimari","year":"2017","journal-title":"Endocrine"},{"issue":"3","key":"2025032012384026300_luaf042-B3","doi-asserted-by":"crossref","first-page":"421","DOI":"10.1016\/j.ecl.2024.05.007","article-title":"Conundrums of diagnosis and management of Cushing's syndrome in pregnancy","volume":"53","author":"Livia Gheorghiu","year":"2024","journal-title":"Endocrinol Metab Clin North Am"},{"issue":"5","key":"2025032012384026300_luaf042-B4","doi-asserted-by":"crossref","first-page":"670","DOI":"10.1007\/s11102-021-01142-7","article-title":"Diagnosis and treatment outcomes of Cushing's disease during pregnancy","volume":"24","author":"Sridharan","year":"2021","journal-title":"Pituitary"},{"issue":"3","key":"2025032012384026300_luaf042-B5","doi-asserted-by":"crossref","first-page":"269","DOI":"10.1007\/s11102-024-01384-1","article-title":"Prolactin-secreting tumors, dopamine agonists and pregnancy: a longitudinal experience of a tertiary neuroendocrine center","volume":"27","author":"Prencipe","year":"2024","journal-title":"Pituitary"},{"issue":"5","key":"2025032012384026300_luaf042-B6","doi-asserted-by":"crossref","first-page":"3077","DOI":"10.1210\/jc.2004-2361","article-title":"Cushing's syndrome during pregnancy: personal experience and review of the literature","volume":"90","author":"Lindsay","year":"2005","journal-title":"J Clin Endocrinol Metab"},{"issue":"2","key":"2025032012384026300_luaf042-B7","doi-asserted-by":"crossref","first-page":"78","DOI":"10.1016\/j.aace.2021.10.004","article-title":"Cushing disease treated successfully with metyrapone during pregnancy","volume":"8","author":"Bass","year":"2021","journal-title":"AACE Clin Case Rep"},{"issue":"1","key":"2025032012384026300_luaf042-B8","doi-asserted-by":"crossref","first-page":"55","DOI":"10.31053\/1853.0605.v80.n1.37056","article-title":"Enfermedad de Cushing en el embarazo y uso de cabergolina. [Cushing's disease in pregnancy and treatment with cabergoline: obstetric and neonatal results]","volume":"80","author":"Murillo","year":"2023","journal-title":"Rev Fac Cien Med Univ Nac Cordoba"},{"issue":"10","key":"2025032012384026300_luaf042-B9","doi-asserted-by":"crossref","first-page":"1003","DOI":"10.1507\/endocrj.EJ23-0725","article-title":"Cushing's disease with twin pregnancy and diabetes mellitus: a case report and literature review","volume":"71","author":"Asai","year":"2024","journal-title":"Endocr J"},{"issue":"4","key":"2025032012384026300_luaf042-B10","doi-asserted-by":"crossref","first-page":"314","DOI":"10.1111\/ajo.13793","article-title":"Diverse presentations of Cushing's syndrome during pregnancy\u2014a case series","volume":"64","author":"Stoinis","year":"2024","journal-title":"Aust N Z J Obstet Gynaecol"},{"issue":"7-8","key":"2025032012384026300_luaf042-B11","first-page":"16528","article-title":"Pulmonary embolism after delivery as the first manifestation of Cushing disease in pregnancy","volume":"133","author":"Minasyan","year":"2023","journal-title":"Pol Arch Intern Med"},{"key":"2025032012384026300_luaf042-B12","doi-asserted-by":"crossref","first-page":"23247096231204732","DOI":"10.1177\/23247096231204732","article-title":"A case report of Cushing's disease presenting with psychosis and muscle weakness postpartum","volume":"11","author":"Eskandari","year":"2023","journal-title":"J Investig Med High Impact Case Rep"},{"issue":"12","key":"2025032012384026300_luaf042-B13","doi-asserted-by":"crossref","first-page":"847","DOI":"10.1016\/S2213-8587(21)00235-7","article-title":"Consensus on diagnosis and management of Cushing's disease: a guideline update","volume":"9","author":"Fleseriu","year":"2021","journal-title":"Lancet Diabetes Endocrinol"},{"issue":"1","key":"2025032012384026300_luaf042-B14","doi-asserted-by":"crossref","first-page":"23","DOI":"10.1007\/s11154-022-09731-y","article-title":"Endogenous Cushing's syndrome during pregnancy","volume":"24","author":"Younes","year":"2023","journal-title":"Rev Endocr Metab Disord"},{"issue":"1","key":"2025032012384026300_luaf042-B15","doi-asserted-by":"crossref","first-page":"30","DOI":"10.1007\/s11102-015-0680-3","article-title":"Determination of nighttime salivary cortisol during pregnancy: comparison with values in non-pregnancy and Cushing's disease","volume":"19","author":"Lopes","year":"2016","journal-title":"Pituitary"},{"issue":"11","key":"2025032012384026300_luaf042-B16","doi-asserted-by":"crossref","first-page":"1227","DOI":"10.1530\/EC-18-0400","article-title":"Preoperative treatment with metyrapone in patients with Cushing's syndrome due to adrenal adenoma","volume":"7","author":"Puglisi","year":"2018","journal-title":"Endocr Connect"},{"key":"2025032012384026300_luaf042-B17","doi-asserted-by":"crossref","first-page":"259","DOI":"10.1007\/978-1-60327-449-4_21","volume-title":"Cushing's Syndrome. Contemporary Endocrinology","author":"Abdelmannan","year":"2010"}],"container-title":["JCEM Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/jcemcr\/article-pdf\/3\/4\/luaf042\/62490653\/luaf042.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/jcemcr\/article-pdf\/3\/4\/luaf042\/62490653\/luaf042.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,3,20]],"date-time":"2025-03-20T23:57:51Z","timestamp":1742515071000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/jcemcr\/article\/doi\/10.1210\/jcemcr\/luaf042\/8087989"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,3,17]]},"references-count":17,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2025,3,17]]}},"URL":"https:\/\/doi.org\/10.1210\/jcemcr\/luaf042","relation":{},"ISSN":["2755-1520"],"issn-type":[{"value":"2755-1520","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2025,4]]},"published":{"date-parts":[[2025,3,17]]},"article-number":"luaf042"}}