{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,1]],"date-time":"2026-09-01T23:33:46Z","timestamp":1788305626874,"version":"build-2803163510"},"reference-count":44,"publisher":"Walter de Gruyter GmbH","issue":"4","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2017,1,1]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Background:<\/jats:title><jats:p>Adenocorticotropic hormone (ACTH)-dependent Cushing\u2019s syndrome in infancy is extremely rare. We describe the case of a sacro-coccygeal ectopic ACTH-secreting immature teratoma in an infant who also presented the triad of defects characteristic of Currarino syndrome.<\/jats:p><\/jats:sec><jats:sec><jats:title>Case presentation:<\/jats:title><jats:p>A girl was born with a large immature teratoma in the sacro-coccygeal region associated with anal atresia. At the age of 7 days, the concentration of \u03b1-fetoprotein (AFP) was above the age-specific normal range. Two non-radical surgical excisions of the tumour were performed. At the age of 7 months, she developed polyphagia, acne, hirsutism, hypertension and hypokalemia with elevated ACTH and absence of serum cortisol circadian rhythm. Immunostaining of tumour tissue showed ACTH-immunoreactive cells. Due to unsuccessful therapy with ketoconazole and resistance to antihypertensive medications [blood pressure (BP) 210\/160 mmHg], metyrapone was administered, which controlled her ACTH and cortisol levels in the normal range. Following further removal of tumour bulk after three operations during the first year of life, there was a decrease of BP to normal values.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions:<\/jats:title><jats:p>A rare case of ectopic ACTH syndrome causing Cushing\u2019s syndrome in infancy in the context of Currarino syndrome is reported. Radical surgery has resulted in excision of the tumour and current control of Cushing\u2019s syndrome.<\/jats:p><\/jats:sec>","DOI":"10.1515\/jpem-2016-0339","type":"journal-article","created":{"date-parts":[[2017,3,22]],"date-time":"2017-03-22T10:04:26Z","timestamp":1490177066000},"source":"Crossref","is-referenced-by-count":7,"title":["Cushing\u2019s syndrome in infancy due to ectopic ACTH secretion by a sacro-coccygeal teratoma"],"prefix":"10.1515","volume":"30","author":[{"given":"Marta","family":"Rydzewska","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maryna","family":"Krawczuk-Rybak","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Adrianna","family":"Zajkowska","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Natalia","family":"Jurczuk","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Dariusz","family":"Polnik","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mieczys\u0142aw","family":"Szalecki","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"El\u017cbieta","family":"Moszczy\u0144ska","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Martin O.","family":"Savage","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Artur","family":"Bossowski","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"374","reference":[{"key":"ref321","doi-asserted-by":"crossref","first-page":"2359","DOI":"10.1002\/1097-0142(197605)37:5<2359::AID-CNCR2820370528>3.0.CO;2-Q","article-title":"Immature (malignant) teratoma of the ovary: a clinical and pathologic study of 58 cases","volume":"37","year":"1976","journal-title":"Cancer"},{"key":"ref391","first-page":"281","article-title":"Cushing\u2019s syndrome: epidemiology and developments in disease management","volume":"7","year":"2015","journal-title":"Clin Epidemiol"},{"key":"ref131","doi-asserted-by":"crossref","first-page":"1043","DOI":"10.1515\/jpem-2014-0230","article-title":"How early can one diagnose Cushing\u2019s disease? 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