{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T03:01:16Z","timestamp":1787022076859,"version":"build-2736575974"},"reference-count":59,"publisher":"Public Library of Science (PLoS)","issue":"11","license":[{"start":{"date-parts":[[2020,11,25]],"date-time":"2020-11-25T00:00:00Z","timestamp":1606262400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["www.plosone.org"],"crossmark-restriction":false},"short-container-title":["PLoS ONE"],"abstract":"<jats:sec id=\"sec001\">\n                    <jats:title>Purpose<\/jats:title>\n                    <jats:p>Ectopic Cushing Syndrome (EAS) is a rare condition responsible for about 5\u201320% of all Cushing syndrome cases. It increases the mortality of affected patients thus finding and removal of the ACTH-producing source allows for curing or reduction of symptoms and serum cortisol levels. The aim of this study is to present a 20-year experience in the diagnosis and clinical course of patients with EAS in a single Clinical Centre in Southern Poland as well as a comparison of clinical course and outcomes depending on the source of ectopic ACTH production\u2013especially neuroendocrine tumors with other neoplasms.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec id=\"sec002\">\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Twenty-four patients were involved in the clinical study with EAS diagnosed at the Department of Endocrinology between years 2000 and 2018. The diagnosis of EAS was based on the clinical presentation, hypercortisolemia with high ACTH levels, high dose dexamethasone suppression test and\/or corticotropin-releasing hormone tests. To find the source of ACTH various imaging studies were performed.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec id=\"sec003\">\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Half of the patients were diagnosed with neuroendocrine tumors, whereby muscle weakness was the leading symptom. Typical cushingoid appearance was seen in merely a few patients, and weight loss was more common than weight gain. Patients with neuroendocrine tumors had significantly higher midnight cortisol levels than the rest of the group. Among patients with infections, we observed a significantly higher concentrations of cortisol 2400 levels in gastroenteropancreatic neuroendocrine tumors. Chromogranin A correlated significantly with potassium in patients with neuroendocrine tumors and there was a significant correlation between ACTH level and severity of hypokalemia.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec id=\"sec004\">\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>EAS is not common, but if it occurs it increases the mortality of patients; therefore, it should be taken into consideration in the case of coexistence of severe hypokalemia with hypertension and muscle weakness, especially when weight loss occurs. Because the diagnosis of gastroenteropancreatic neuroendocrine tumor worsens the prognosis-special attention should be paid to these patients.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1371\/journal.pone.0242679","type":"journal-article","created":{"date-parts":[[2020,11,25]],"date-time":"2020-11-25T13:30:58Z","timestamp":1606311058000},"page":"e0242679","update-policy":"https:\/\/doi.org\/10.1371\/journal.pone.corrections_policy","source":"Crossref","is-referenced-by-count":34,"title":["Ectopic ACTH syndrome of different origin\u2014Diagnostic approach and clinical outcome. Experience of one Clinical Centre"],"prefix":"10.1371","volume":"15","author":[{"given":"Joanna Ewelina","family":"Pale\u0144-Tytko","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0936-1403","authenticated-orcid":true,"given":"Elwira Maria","family":"Przybylik-Mazurek","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ewelina Joanna","family":"Rzepka","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dorota Magdalena","family":"Pach","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna Stanis\u0142awa","family":"Sowa-Staszczak","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aleksandra","family":"Gilis-Januszewska","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alicja Bronis\u0142awa","family":"Hubalewska-Dydejczyk","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"340","published-online":{"date-parts":[[2020,11,25]]},"reference":[{"issue":"6","key":"pone.0242679.ref001","first-page":"480","article-title":"Zesp\u00f3\u0142 Cushinga w przebiegu ektopowego wydzielania ACTH","volume":"64","author":"\u0141 Cieszy\u0144ski","year":"2015","journal-title":"Endokrynol Pol"},{"issue":"9996","key":"pone.0242679.ref002","doi-asserted-by":"crossref","first-page":"913","DOI":"10.1016\/S0140-6736(14)61375-1","article-title":"Cushing\u2019s syndrome","volume":"386","author":"A Lacroix","year":"2015","journal-title":"Lancet (London, England)"},{"issue":"8","key":"pone.0242679.ref003","doi-asserted-by":"crossref","first-page":"4955","DOI":"10.1210\/jc.2004-2527","article-title":"Cushing\u2019s Syndrome Due to Ectopic Corticotropin Secretion: Twenty Years\u2019 Experience at the National Institutes of Health","volume":"90","author":"I Ilias","year":"2005","journal-title":"J Clin Endocrinol Metab"},{"issue":"8","key":"pone.0242679.ref004","doi-asserted-by":"crossref","first-page":"1217","DOI":"10.1590\/S0004-27302007000800007","article-title":"Ectopic ACTH syndrome","volume":"51","author":"AM Isidori","year":"2007","journal-title":"Arq Bras Endocrinol Metabol"},{"issue":"9522","key":"pone.0242679.ref005","doi-asserted-by":"crossref","first-page":"1605","DOI":"10.1016\/S0140-6736(06)68699-6","article-title":"Cushing\u2019s syndrome","volume":"367","author":"J Newell-Price","year":"2006","journal-title":"Lancet (London, England)"},{"issue":"1","key":"pone.0242679.ref006","first-page":"117","article-title":"Incidence and late prognosis of cushing\u2019s syndrome: a population-based study","volume":"86","author":"J Lindholm","year":"2001","journal-title":"J Clin Endocrinol Metab"},{"issue":"2","key":"pone.0242679.ref007","first-page":"440","article-title":"Diagnosis and management of Cushing\u2019s syndrome: results of an Italian multicentre study. 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