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Case Report

Feminizing Adrenocortical Carcinoma in Men: A Rare Cause of Persistent Gynecomastia and a Contemporary Literature Review

by
Ana Maria Arnautu
1,†,
Diana Loreta Paun
1,2,*,†,
Corina Neamtu
3,†,
Costin Gingu
4,†,
Victor Nimigean
2,†,
Dana-Mihaela Tilici
1,5,†,
Ruxandra Costinescu
1,†,
Mirona Costea
1,5,†,
Adina Onofrei
1,†,
Beatrice Grecu
1,†,
Claudia Nacea-Radu
1,† and
Sorin Paun
2,6,†
1
Endocrinology Department, Bucharest University Emergency Hospital, 050098 Bucharest, Romania
2
Faculty of Medicine, University of Medicine and Pharmacy “Carol Davila”, 020021 Bucharest, Romania
3
Clinic Hospital SANADOR, 010997 Bucharest, Romania
4
“Fundeni” Clinical Institute, 022328 Bucharest, Romania
5
Doctoral School, University of Medicine and Pharmacy “Carol Davila”, 020021 Bucharest, Romania
6
Bucharest Clinical Emergency Hospital, 014461 Bucharest, Romania
*
Author to whom correspondence should be addressed.
These authors contributed equally to this work.
Epidemiologia 2025, 6(4), 64; https://doi.org/10.3390/epidemiologia6040064
Submission received: 2 August 2025 / Revised: 10 September 2025 / Accepted: 10 October 2025 / Published: 15 October 2025

Abstract

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 µg/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.
Keywords: feminizing adrenocortical carcinoma; bilateral gynecomastia; estrogen-secreting tumor; fertility; low-risk ACC feminizing adrenocortical carcinoma; bilateral gynecomastia; estrogen-secreting tumor; fertility; low-risk ACC

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MDPI and ACS Style

Arnautu, A.M.; Paun, D.L.; Neamtu, C.; Gingu, C.; Nimigean, V.; Tilici, D.-M.; Costinescu, R.; Costea, M.; Onofrei, A.; Grecu, B.; et al. Feminizing Adrenocortical Carcinoma in Men: A Rare Cause of Persistent Gynecomastia and a Contemporary Literature Review. Epidemiologia 2025, 6, 64. https://doi.org/10.3390/epidemiologia6040064

AMA Style

Arnautu AM, Paun DL, Neamtu C, Gingu C, Nimigean V, Tilici D-M, Costinescu R, Costea M, Onofrei A, Grecu B, et al. Feminizing Adrenocortical Carcinoma in Men: A Rare Cause of Persistent Gynecomastia and a Contemporary Literature Review. Epidemiologia. 2025; 6(4):64. https://doi.org/10.3390/epidemiologia6040064

Chicago/Turabian Style

Arnautu, Ana Maria, Diana Loreta Paun, Corina Neamtu, Costin Gingu, Victor Nimigean, Dana-Mihaela Tilici, Ruxandra Costinescu, Mirona Costea, Adina Onofrei, Beatrice Grecu, and et al. 2025. "Feminizing Adrenocortical Carcinoma in Men: A Rare Cause of Persistent Gynecomastia and a Contemporary Literature Review" Epidemiologia 6, no. 4: 64. https://doi.org/10.3390/epidemiologia6040064

APA Style

Arnautu, A. M., Paun, D. L., Neamtu, C., Gingu, C., Nimigean, V., Tilici, D.-M., Costinescu, R., Costea, M., Onofrei, A., Grecu, B., Nacea-Radu, C., & Paun, S. (2025). Feminizing Adrenocortical Carcinoma in Men: A Rare Cause of Persistent Gynecomastia and a Contemporary Literature Review. Epidemiologia, 6(4), 64. https://doi.org/10.3390/epidemiologia6040064

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