Background: Sertoli–Leydig cell tumors (SLCT) are rare ovarian tumor that belongs to the group of sex-cord stromal tumors. These constitute less than 0.5% of all ovarian tumors. Most of these (97%) tumors are unilateral, with bilaterality being very rare. Case: Here we report a very unusual case of bilateral SLCT in association with a unilateral dermoid cyst in a 37-year-old female. She presented with a history of abdominal pain, and bilateral abdominal masses. Ultrasound examination revealed bilateral ovarian masses for which a bilateral salphigo-oopherectomy was performed. The histopathological examination revealed a bilateral Sertoli–Leydig cell tumor associated with a unilateral dermoid cyst. Conclusions: This case emphasizes the varied histopathological morphology of an ovarian neoplasm. (J GYNECOL SURG 29:33)
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