Virilizing ovarian tumors in postmenopause: a literature review and a rare clinical case

DOI

https://dx.doi.org/10.18565/aig.2021.1.178-185

Ponomareva Yu.N., Loginova E.A., Telezhnikova I.M.
A.S. Loginov Moscow Clinical Research and Practical Center, Moscow Healthcare Department, Moscow, Russia

Background. Sertoli–Leydig cell ovarian tumors are rare and account for less than 0.5% of all ovarian tumors. Sertoli–Leydig cell tumors can develop in women of any age group; however, they are more common in young female patients with a mean age of 25 years. The distinguishing feature of Sertoli–Leydig cell tumors is their ability to secrete androgens that may cause defeminization. It is rather difficult to diagnose androgen-producing tumors in postmenopause, since the sharp decline in estrogen levels and a slower decrease in androgen levels while maintaining the synthesis throughout life are natural hormonal changes.
Case report. The paper analyzes literature data and a rare clinical case of Sertoli–Leydig cell tumor in postmenopausal female patient with breast cancer. The patient was observed to have pronounced signs of virilism: hirsutism, deepening voice, alopecia, and clitoromegaly. MRI revealed a 33-mm left solid ovarian tumor accumulating a contrast agent. She was diagnosed with an increase in blood testosterone levels up to 10 nmol/L (normal value, less than 1.24 nmol/L). The patient underwent surgical treatment: laparoscopy, hysterectomy, bilateral removal of the appendages, and omentectomy. Morphological examination diagnosed a moderate left Sertoli–Leydig cell ovarian tumor. The testosterone level was noted to decrease after surgery. No tumor recurred during one year of follow-up.
Conclusion. Thus, postmenopausal patients with pronounced signs of hyperandrogenism should be examined to exclude rare diseases, such as androgen-producing tumors.

Sertoli–Leydig cell tumor

sex cord tumor

postmenopause

defeminization

virilization

androgen-producing tumors

hysterectomy

Full text (in Russian)

Tandon R., Goel P., Saha P.K., Takkar N., Punia R.P. A rare ovarian tumor – Sertoli–Leydig cell tumor with heterologous element. MedGenMed. 2007; 9(4): 44.
Chakrabarti I., De A., Gangopadhyay M., Bera P. Sertoli–Leydig cell tumour of ovary with heterologous elements – a case report. Internet J. Gynecol. Obstet. 2010; 13(1): 1-4.
Ray-Coquard I. Ovarian tumors of sex cord-stromal origin. Orphanet Encyclopedia. 2004: 1-4. Available at: http://www.orpha.net/data/patho/GB/uk-STROMA.pdf
Vaishali D.R., Kalpana D.S., Meeta M.H. Sertoli–Leydig cell tumors of ovary. J. Obstet. Gynecol. (India). 2009; 59(2): 165-7.
Monappa V., Reddy S.M., Kudva R., Pai M.V. Cystic Sertoli–Leydig cell tumour in a postmenopausal woman with absent virilising symptoms: a diagnostic challenge. J. Clin. Diagn. Res. 2017; 11(5): ED26-8. https://dx.doi.org/ 10.7860/JCDR/2017/26173.9901.
Roth L.M., Anderson M.C., Govan A.D.T., Langley F.A., Gowing N.F.C., Woodcock A.S. Sertoli–Leydig cell tumour: a clinicopathologic study of 34 cases. Cancer. 1981; 48(1): 187-97. https://dx.doi.org/10.1002/1097-0142(19810701)48:1<187::aid-cncr2820480130>3.0.co;2-1.
Olt G., Mortel R. Hormone-producing tumors of the ovary. Endocrine-Related Cancer. 1997; 4(4): 447-57. https://dx.doi.org/10.1677/erc.0.0404447.
Черепанова Е.В., Лактионов К.П., Анурова О.А., Толокнов Б.О., Зотиков А.И. Клинико-морфологические особенности опухолей яичников из клеток Сертоли–Лейдига. Опухоли женской репродуктивной системы. 2010; 4: 83-9. [Cherepanova E.V., Laktionov K.P., Anurova O.A., Toloknov B.O., Zotikov A.I. Clinical and morphological features of ovarian tumors from Sertoli–Leydig cells. Tumors of the female reproductive system. 2010; 4: 83-9.(in Russian)].
Gouy S., Arfi A., Maulard A., Pautier P., Bentivegna E., Leary A. et al. Results from a monocentric long-term analysis of 23 patients with ovarian Sertoli–Leydig cell tumors. Oncologist. 2019; 24(5): 702-9. https://dx.doi.org/10.1634/theoncologist.2017-0632.
Omeroglu A., Husain A.N., Siziopikou K. Pathologic quiz case: a papillary ovarian tumor in a 4-year-old girl. Arch. Pathol. Lab. Med. 2002; 126(3): 377-8. https://dx.doi.org/10.1043/0003-9985(2002)126<0377:PQCAPO>2.0.CO;2.
Chen M., Zhou W., Zhang Z., Zou Y., Li C. An ovarian Leydig cell tumor of ultrasound negative in a postmenopausal woman with hirsutism and hyperandrogenism: a case report. Medicine (Baltimore). 2018; 97(10): e0093. https://dx.doi.org/10.1097/MD.0000000000010093.
Young R.H. Sex cord-stromal tumors of the ovary and testis: their similarities and differences with consideration of selected problems. Mod. Pathol. 2005; 18(Suppl. 2): S81-98. https://dx.doi.org/10.1038/modpathol.3800311.
Adashi E.Y. The climacteric ovary as a functional gonadotropin-driven androgen-producing gland. Fertil. Steril. 1994; 62(1): 20-7.https://dx.doi.org/10.1016/s0015-0282(16)56810-1.
Seidman J.D., Patterson J.A., Bitterman P. Sertoli–Leydig cell tumor associated with a mature cystic teratoma in a single ovary. Mod. Pathol. 1989; 2(6): 687-92.
Gautam P., Rao M., Gothwal M., Garg P.K., Bhattacharya S. Sertoli–Leydig cell tumor of ovary: a rare case report with heterologous elements and focal marked anaplasia. Int. J. Appl. Basic Med. Res. 2019; 9(1): 62-4. https://dx.doi.org/10.4103/ijabmr.IJABMR_84_18.
Burnik Papler T., Frković Grazio S., Kobal B. Sertoli–Leydig cell tumor with retiform areas and overgrowth of rhabdomyosarcomatous elements: case report and literature review. J. Ovarian Res. 2016; 9(1): 46. https://dx.doi.org/10.1186/s13048-016-0257-4.
Sherf S., Martinez D. Leydig cell tumor in the post-menopausal woman: case report and literature review. Acta Biomed. 2016; 87(3): 310-3.
Moghazy D., Sharan C., Nair M., Rackauskas C., Burnette R., Diamond M. et al. Sertoli–Leydig cell tumor with unique nail findings in a post-menopausal woman: a case report and literature review. J. Ovarian Res. 2014; 7: 83. https://dx.doi.org/10.1186/s13048-014-0083-5.
Inam Z.S., Azizi A.H., Holland S.W. An 88-year-old woman with flushing, alopecia and hirsutism and a Sertoli–Leydig cell tumour. BMJ Case Rep. 2018; 2018: bcr2017222589. https://dx.doi.org/10.1136/bcr-2017-222589.
Tsuzuki Y., Kikuchi I., Nojima M., Yoshida K., Hashizume A., Tomita S. A case report: ovarian Sertoli–Leydig cell tumor with hyperestrogenism and endometrial hyperplasia in a postmenopausal woman. Jpn. Clin. Med. 2017; 8: 1179066017695239. https://dx.doi.org/10.1177/1179066017695239.
Brandone N., Borrione C., Rome A., Maues de Paula A. Ovarian Sertoli–Leydig tumor: a tricky tumor. Ann. Pathol. 2018; 38(2): 131-6. https://dx.doi.org/10.1016/j.annpat.2018.01.002.
Moussa D., Aziz D.A., Niassy D.A., Espérence K.C., Youssou N., Charles M.J. et al. Sertoli-Leydig cell tumor of the ovary: case study of a 22-year old woman. Pan. Afr. Med. J. 2016; 25: 36. https://dx.doi.org/10.11604/pamj.2016.25.36.10469.
Turkyilmaz Z., Karabulut R., Ozbakir F., Pinarli G., Arda Inan M., Oguz A. et al. Recurrent Sertoli–Leydig cell tumor of ovary in 8-year-old girl. Scott. Med. J. 2016; 61(1): 56-9. https://dx.doi.org/10.1177/0036933015619297.
Stavrakis T., Kalogiannidis I., Petousis S., Tsompanidou C., Delkos D., Prapas N. et al. Fertility-sparing management and obstetric outcomes in a 20-year-old patient with a Sertoli–Leydig cell tumor of the ovary: a case report and review of the literature. Oncol. Lett. 2016; 12(2): 1079-82. https://dx.doi.org/10.3892/ol.2016.4695.
Liggins C.A., Ma L.T., Schlumbrecht M.P. Sertoli–Leydig cell tumor of the ovary: a diagnostic dilemma. Gynecol. Oncol. Rep. 2015; 15: 16-9. https://dx.doi.org/10.1016/j.gore.2015.12.003.
Zhang H., Hao J., Li C.Y., Li T., Mu Y.L. Large moderately-differentiated ovarian Sertoli–Leydig cell tumor in a 13-year-old female: a case report. Oncol. Lett. 2016; 11(2): 1110-2. https://dx.doi.org/10.3892/ol.2015.3979.
Zhang H.Y., Zhu J.E., Huang W., Zhu J. Clinicopathologic features of ovarian Sertoli-Leydig cell tumors. Int. J. Clin. Exp. Pathol. 2014; 7(10): 6956-64.
Rohini Dhanya C.S., Padhi S., Varghese R.G. Sertoli–Leydig cell tumor of ovary – a diagnostic dilemma. J. Clin. Diagn. Res. 2014; 8(3): 127-9. https://dx.doi.org/10.7860/JCDR/2014/7000.4099.
Chen L., Tunnell C.D., De Petris G. Sertoli–Leydig cell tumor with heterologous element: a case report and a review of the literature. Int. J. Clin. Exp. Pathol. 2014; 7(3): 1176-81.
Perez Lana M., Demayo S., Monastero A., Nolting M. Ovarian tumors secreting androgens: an infrequent cause of hyperandrogenism. Minerva Ginecol. 2019; 71(1): 72-7. https://dx.doi.org/10.23736/S0026-4784.18.04302-2.
Xu Q., Zou Y., Zhang X.F. Sertoli-Leydig cell tumors of ovary: a case series. Medicine (Baltimore). 2018; 97(42): e12865. https://dx.doi.org/ 10.1097/MD.0000000000012865.
Akman L., Ertas I.E., Gokcu M., Terek M.C., Sanci M., Sanli U.A. et al. Ovarian sertoli-leydig cell tumors: a multicenter long-term clinicopathological analysis of 27 patients. J. Cancer Res. Ther. 2016; 12(1): 290-4. https://dx.doi.org/10.4103/0973-1482.158037.
Schneider D.T., Orbach D., Cecchetto G., Stachowicz-Stencel T., Brummel B.,Brecht I.B. et al. Ovarian Sertoli–Leydig cell tumours in children and adolescents: an analysis of the European Cooperative Study Group on Pediatric Rare Tumors (EXPeRT). Eur. J. Cancer. 2015; 51(4): 543-50. https://dx.doi.org/10.1016/j.ejca.2014.11.013.
Young R.H., Scully R.E. Ovarian Sertoli cell tumors: a report of 10 cases. Int. J. Gynecol. Pathol. 1984; 2(4): 349-63. https://dx.doi.org/10.1097/00004347-198404000-00003.
Gressel G.M., Buza N., Pal L. Ovarian Sertoli–Leydig cell tumors: a single institution experience and review of the literature. Eur. J. Gynaecol. Oncol. 2017; 38(2): 214-20.
Oliva E., Alvarez T., Young R.H. Sertoli cell tumors of the ovary: a clinicopathological and immunohistochemical study of 54 cases. Am. J. Surg. Pathol. 2005; 29(2): 143-56. https://dx.doi.org/10.1097/01.pas.0000149692.21205.9c.
Weng C.S., Chen M.Y., Wang T.Y., Tsai H.W., Hung Y.C., Yu K.J. et al. Sertoli–Leydig cell tumors of the ovary: a Taiwanese Gynecologic Oncology Group study. Taiwan. J. Obstet. Gynecol. 2013; 52(1): 66-70. https://dx.doi.org/10.1016/j.tjog.2012.03.001.
Gui T., Cao D., Shen K., Yang J., Zhang Y., Yu Q. et al. A clinicopathological analysis of 40 cases of ovarian Sertoli–Leydig cell tumors. Gynecol. Oncol. 2012; 127(2): 384-9. https://dx.doi.org/10.1016/j.ygyno.2012.07.114.
Хохлова С.В., Давыдова И.Ю., Коломиец Л.А., Кузнецов В.В., Новикова Е.Г., Трякин А.А., Тюляндина А.С., Урманчеева А.Ф. Практические рекомендации по лекарственному лечению злокачественных неэпителиальных опухолей яичников. Злокачественные опухоли. 2019; 9(3, Приложение 2): 177-91. [Hokhlova S.V, Davydova I.Yu., Kolomiets L.A., Kuznetsov V.V., Novikova E.G., Tryakin A.A., Tyulyandina A.S., Urmancheeva A.F. Practical recommendations for the drug treatment of malignant non-epithelial ovarian tumors. Malignant tumor. 2019; 9(3, Suppl. 2): 177-91. (in Russian)]. https://dx.doi.org/10.18027 / 2224-5057-2019-9-3s2-177-191.

Received 16.09.2020

Accepted 11.12.2020

Yulia N. Ponomareva, MD, PhD, Professor, Head of the Department of Gynecology, Moscow Clinical Scientific Center, Moscow Healthcare Department.
Tel.: +7(495)304-30-35. E-mail: y.ponomareva@mknc.ru. ORCID: 0000-0001-5436-9119.
11123, Russia, Moscow, shosse Entuziastov, 86.
Ekaterina A. Loginova, MD, Research Fellow of the Department of Gynecology, Moscow Clinical Scientific Center, Moscow Healthcare Department.
Tel.: +7 (495)304-30-35; +7(985)182-28-58. E-mail: e.loginova@mknc.ru. ORCID: 0000-0002-5061-1227.
11123, Russia, Moscow, shosse Entuziastov, 86.
Inessa M. Telezhnikova, Research Fellow of the Department of Morbid Anatomy, Moscow Clinical Scientific Center, Moscow Healthcare Department.
Tel.: +7 (495)304-30-35; +7(909)956-33-39. E-mail: inessssa@inbox.ru. ORCID: 0000-0002-1491-2882.
11123, Russia, Moscow, shosse Entuziastov, 86.

For citation: Ponomareva Yu.N., Loginova E.A., Telezhnikova I.M. Virilizing ovarian tumors in postmenopause: a literature review and a rare clinical case.
Akusherstvo i Ginekologiya/Obstetrics and Gynecology. 2021; 1: 178-185 (in Russian)
https://dx.doi.org/10.18565/aig.2021.1.178-185

Virilizing ovarian tumors in postmenopause: a literature review and a rare clinical case

Ponomareva Yu.N., Loginova E.A., Telezhnikova I.M.

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