Objectives We reported a Sertoli-Leydig cell tumor with symptoms of hyperandrogenism.

Methods Sertoli-Leydig cell tumor (SLCT) also called androblastoma represents less than 0,5% of all primary ovarian tumor. The majority of SLCTs are unilateral and confined to the ovaries. It’s more common in young women between the age of 25 and 35. In approximately 90% of the cases are diagnosed as Stage IA.

Results We reported a case of SLCT in a 17-year-old female who was presented at emergency room with acute abdominal pain caused by an ovarian torsion. She underwent an emergency laparotomy with confirmed finding of an ovarian tumor without disease at peritoneal cavity. Histopathological examination showed a well encapsulated gray colored solid mass with smooth external surface measuring 16.0 x 14.5 x 11.0 cm. The patient did not receive any adjuvant treatment. After ten months, she presented a pelvic peritoneal recurrence associated with symptoms of hyperandrogenism including hirsutism, deepening of the voice as well amenorrhea. Optimal oncologic cytoreduction surgery was performed. Histopathological exam confirmed Sertoli- Leydig tumor. Immunohistochemical revealed positive for inhibin alpha, FOXL2 and calretinin. The patient received six cycles of carboplatin and paclitaxel chemotherapy regimen. After treatment, the symptoms of hyperandrogenism disappeared. In the moment, the patient has been in a follow-up without any evidence of disease.

Conclusions There is not consensus about the best treatment options. Surgery is still the standard treatment of primary and resectable cases. After recurrence, chemotherapy with platinum and taxares agents are useful in clinical practice.