{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,19]],"date-time":"2026-07-19T18:01:57Z","timestamp":1784484117281,"version":"3.55.0"},"reference-count":8,"publisher":"BMJ","issue":"2","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Rep"],"accepted":{"date-parts":[[2025,11,7]]},"published-print":{"date-parts":[[2026,2]]},"abstract":"<jats:p>Ectopic Cushing syndrome (ECS) occurs when a non-pituitary tumour secretes excess adrenocorticotropic hormone, leading to hypercortisolism. ECS is most commonly associated with small cell lung cancer and pancreatic neuroendocrine tumours, but rarely with oesophageal neuroendocrine carcinoma (NEC). We report the case of a man in the 60s initially treated for locally advanced large cell NEC of the oesophagus who later developed ECS. Oesophageal NECs account for less than 0.04% of all neuroendocrine tumours and carry a poor prognosis, with large cell subtype being particularly uncommon. Despite endocrine-directed therapy, the condition of the patient deteriorated rapidly due to aggressive disease. This case underscores the diagnostic challenges of ECS in rare tumour types and highlights the importance of early recognition and multidisciplinary management to optimise outcomes.<\/jats:p>","DOI":"10.1136\/bcr-2025-265632","type":"journal-article","created":{"date-parts":[[2026,2,4]],"date-time":"2026-02-04T14:55:52Z","timestamp":1770216952000},"page":"e265632","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":0,"title":["Ectopic ACTH production from oesophageal neuroendocrine carcinoma causing Cushing syndrome"],"prefix":"10.1136","volume":"19","author":[{"given":"Yifei","family":"Zhang","sequence":"first","affiliation":[{"name":"Cancer, Smilow Cancer Hospital at Yale New Haven, New Haven, CT, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xuchen","family":"Zhang","sequence":"additional","affiliation":[{"name":"Pathology, Yale University, New Haven, CT, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sachin","family":"Majumdar","sequence":"additional","affiliation":[{"name":"Endocrinology, Yale University Department of Endocrinology and Metabolism, New Haven, CT, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"239","published-online":{"date-parts":[[2026,2,4]]},"reference":[{"key":"2026071910151446000_19.2.e265632.1","doi-asserted-by":"crossref","first-page":"347","DOI":"10.1016\/j.tem.2023.12.003","article-title":"A clinical perspective on ectopic Cushing\u2019s syndrome","volume":"35","author":"Ragnarsson","year":"2024","journal-title":"Trends Endocrinol Metab"},{"key":"2026071910151446000_19.2.e265632.2","doi-asserted-by":"publisher","DOI":"10.21037\/jtd.2016.04.21"},{"key":"2026071910151446000_19.2.e265632.3","doi-asserted-by":"crossref","first-page":"1520","DOI":"10.7150\/jca.29483","article-title":"A 10-year Population-based Study of the Differences between NECs and Carcinomas of the Esophagus in Terms of Clinicopathology and Survival","volume":"10","author":"Cai","year":"2019","journal-title":"J Cancer"},{"key":"2026071910151446000_19.2.e265632.4","doi-asserted-by":"crossref","DOI":"10.1530\/EO-23-0002","article-title":"Ectopic ACTH Cushing\u2019s syndrome caused by a large-cell neuroendocrine lung carcinoma responding to desmopressin","volume":"3","author":"Larose","year":"2023","journal-title":"Endocr Oncol"},{"key":"2026071910151446000_19.2.e265632.5","doi-asserted-by":"publisher","DOI":"10.1159\/000443172"},{"key":"2026071910151446000_19.2.e265632.6","doi-asserted-by":"crossref","first-page":"404","DOI":"10.1159\/000502825","article-title":"Chemotherapy in Resected Neuroendocrine Carcinomas of the Digestive Tract: A National Study from the French Group of Endocrine Tumours","volume":"110","author":"Pellat","year":"2020","journal-title":"Neuroendocrinology"},{"key":"2026071910151446000_19.2.e265632.7","doi-asserted-by":"crossref","first-page":"1721","DOI":"10.1245\/s10434-015-5013-2","article-title":"Surgical Treatment as a Principle for Patients with High-Grade Pancreatic Neuroendocrine Carcinoma: A Nordic Multicenter Comparative Study","volume":"23","author":"Haugvik","year":"2016","journal-title":"Ann Surg Oncol"},{"key":"2026071910151446000_19.2.e265632.8","doi-asserted-by":"publisher","DOI":"10.1530\/EJE-19-0877"}],"container-title":["BMJ Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/syndication.highwire.org\/content\/doi\/10.1136\/bcr-2025-265632","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,7,19]],"date-time":"2026-07-19T17:15:22Z","timestamp":1784481322000},"score":1,"resource":{"primary":{"URL":"https:\/\/casereports.bmj.com\/lookup\/doi\/10.1136\/bcr-2025-265632"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,2]]},"references-count":8,"journal-issue":{"issue":"2","published-online":{"date-parts":[[2026,2,4]]},"published-print":{"date-parts":[[2026,2]]}},"alternative-id":["10.1136\/bcr-2025-265632"],"URL":"https:\/\/doi.org\/10.1136\/bcr-2025-265632","relation":{},"ISSN":["1757-790X"],"issn-type":[{"value":"1757-790X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,2]]}}}