{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,2]],"date-time":"2026-10-02T04:24:33Z","timestamp":1790915073885,"version":"4.1.0"},"reference-count":30,"publisher":"MDPI AG","issue":"4","license":[{"start":{"date-parts":[[2025,10,15]],"date-time":"2025-10-15T00:00:00Z","timestamp":1760486400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Epidemiologia"],"abstract":"<jats:p>Background: Feminizing adrenocortical tumors (FATs) are an exceedingly rare subset of adrenal neoplasms, typically affecting adult men and characterized by an excess of estrogen, suppressed gonadotropins, and gynecomastia. Most FATs are malignant, with a poor prognosis and a high risk of recurrence. Case Presentation: We report the case of a 24-year-old male with bilateral gynecomastia, abdominal mass symptoms, and one year of unexplained infertility. A hormonal evaluation revealed elevated estradiol (90.1 pg\/mL) and suppressed ACTH (2.6 pg\/mL), with inappropriately normal cortisol levels (12.1 \u00b5g\/dL). Imaging identified a right adrenal mass. The patient underwent open adrenalectomy, and histopathology confirmed stage II adrenocortical carcinoma (T2NxM0) with autonomous estradiol secretion, negative margins, and a Ki-67 index of 10%. Postoperatively, gonadal function normalized, and infertility resolved at two months. The multidisciplinary tumor board considered but did not initiate adjuvant mitotane, given the completely resected low-stage disease. Conclusions: This case illustrates the rare presentation of feminizing adrenocortical carcinoma with reversible infertility and highlights the importance of early recognition and close surveillance. In addition, our literature review of 12 male cases reported between 2015 and 2025 emphasizes gynecomastia as the hallmark presentation and discusses emerging evidence supporting active surveillance as a potential alternative to adjuvant mitotane in selected low-risk patients.<\/jats:p>","DOI":"10.3390\/epidemiologia6040064","type":"journal-article","created":{"date-parts":[[2025,10,15]],"date-time":"2025-10-15T08:34:26Z","timestamp":1760517266000},"page":"64","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Feminizing Adrenocortical Carcinoma in Men: A Rare Cause of Persistent Gynecomastia and a Contemporary Literature Review"],"prefix":"10.3390","volume":"6","author":[{"given":"Ana Maria","family":"Arnautu","sequence":"first","affiliation":[{"name":"Endocrinology Department, Bucharest University Emergency Hospital, 050098 Bucharest, Romania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Diana Loreta","family":"Paun","sequence":"additional","affiliation":[{"name":"Endocrinology Department, Bucharest University Emergency Hospital, 050098 Bucharest, Romania"},{"name":"Faculty of Medicine, University of Medicine and Pharmacy \u201cCarol Davila\u201d, 020021 Bucharest, Romania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Corina","family":"Neamtu","sequence":"additional","affiliation":[{"name":"Clinic Hospital SANADOR, 010997 Bucharest, Romania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Costin","family":"Gingu","sequence":"additional","affiliation":[{"name":"\u201cFundeni\u201d Clinical Institute, 022328 Bucharest, Romania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6056-3214","authenticated-orcid":false,"given":"Victor","family":"Nimigean","sequence":"additional","affiliation":[{"name":"Faculty of Medicine, University of Medicine and Pharmacy \u201cCarol Davila\u201d, 020021 Bucharest, Romania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0001-5655-2694","authenticated-orcid":false,"given":"Dana-Mihaela","family":"Tilici","sequence":"additional","affiliation":[{"name":"Endocrinology Department, Bucharest University Emergency Hospital, 050098 Bucharest, Romania"},{"name":"Doctoral School, University of Medicine and Pharmacy \u201cCarol Davila\u201d, 020021 Bucharest, Romania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ruxandra","family":"Costinescu","sequence":"additional","affiliation":[{"name":"Endocrinology Department, Bucharest University Emergency Hospital, 050098 Bucharest, Romania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mirona","family":"Costea","sequence":"additional","affiliation":[{"name":"Endocrinology Department, Bucharest University Emergency Hospital, 050098 Bucharest, Romania"},{"name":"Doctoral School, University of Medicine and Pharmacy \u201cCarol Davila\u201d, 020021 Bucharest, Romania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Adina","family":"Onofrei","sequence":"additional","affiliation":[{"name":"Endocrinology Department, Bucharest University Emergency Hospital, 050098 Bucharest, Romania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Beatrice","family":"Grecu","sequence":"additional","affiliation":[{"name":"Endocrinology Department, Bucharest University Emergency Hospital, 050098 Bucharest, Romania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Claudia","family":"Nacea-Radu","sequence":"additional","affiliation":[{"name":"Endocrinology Department, Bucharest University Emergency Hospital, 050098 Bucharest, Romania"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0007-8301-4131","authenticated-orcid":false,"given":"Sorin","family":"Paun","sequence":"additional","affiliation":[{"name":"Faculty of Medicine, University of Medicine and Pharmacy \u201cCarol Davila\u201d, 020021 Bucharest, Romania"},{"name":"Bucharest Clinical Emergency Hospital, 014461 Bucharest, Romania"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2025,10,15]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"651","DOI":"10.4081\/cp.2014.651","article-title":"Feminizing Adrenocortical Carcinoma with Distant Metastases: Can Surgery Be Considered?","volume":"4","author":"Fancellu","year":"2014","journal-title":"Clin. 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