{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,17]],"date-time":"2026-09-17T23:04:00Z","timestamp":1789686240728,"version":"4.0.1"},"reference-count":24,"publisher":"The Endocrine Society","issue":"2","license":[{"start":{"date-parts":[[2025,1,20]],"date-time":"2025-01-20T00:00:00Z","timestamp":1737331200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,1,20]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Cushing syndrome due to ectopic adrenocorticotropic hormone (ACTH) secretion (EAS) is rare and may progress rapidly, making treatment very challenging. We report a 27-year-old woman with metastatic neuroendocrine tumor (NET) who presented with sudden onset and rapidly progressing fatigue, muscle weakness, and weight gain. Laboratory findings confirmed severe EAS with new onset hypocalcemia, hypokalemia, and hyperglycemia. The patient was hospitalized and initiated on high-dose osilodrostat. Cortisol levels rapidly decreased in response to osilodrostat. During treatment, she developed glucocorticoid withdrawal symptoms for which hydrocortisone treatment was started. Due to her ineligibility for bilateral adrenalectomy, bilateral adrenal arterial embolization was attempted but ultimately converted to right-only embolization due to hypertensive urgency and difficulty in catheterization. With significant improvement of cortisol levels, her hypocalcemia, hyperglycemia, and hypokalemia resolved. Her osilodrostat dose was gradually lowered during follow-up. Glucocorticoid replacement continued as part of the ongoing block-and-replace regimen.<\/jats:p>","DOI":"10.1210\/jcemcr\/luae255","type":"journal-article","created":{"date-parts":[[2025,1,20]],"date-time":"2025-01-20T08:21:17Z","timestamp":1737361277000},"source":"Crossref","is-referenced-by-count":6,"title":["Successful Treatment of Severe Ectopic ACTH-Dependent Cushing Syndrome Complicated by Hypocalcemia With Osilodrostat"],"prefix":"10.1210","volume":"3","author":[{"ORCID":"https:\/\/orcid.org\/0009-0004-3395-8458","authenticated-orcid":false,"given":"Kristen","family":"Lee","sequence":"first","affiliation":[{"name":"Division of Endocrinology, Metabolism and Molecular Medicine, Northwestern University Feinberg School of Medicine , Chicago, IL 60611 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carolina","family":"Mendes Pessoa","sequence":"additional","affiliation":[{"name":"Division of Endocrinology, Metabolism and Molecular Medicine, Northwestern University Feinberg School of Medicine , Chicago, IL 60611 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7329-1887","authenticated-orcid":false,"given":"Wenyu","family":"Huang","sequence":"additional","affiliation":[{"name":"Division of Endocrinology, Metabolism and Molecular Medicine, Northwestern University Feinberg School of Medicine , Chicago, IL 60611 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"80","published-online":{"date-parts":[[2025,1,20]]},"reference":[{"issue":"4","key":"2025020513062114700_luae255-B1","first-page":"23","article-title":"Severe Cushing's syndrome from an ectopic adrenocorticotropic hormone-secreting neuroendocrine tumour treated by osilodrostat","volume":"2023","author":"H\u00e1na","year":"2023","journal-title":"Endocrinol Diabetes Metab Case 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