Ovarian Sertoli-Leydig cell tumor in an amenorrheic hirsute patient.

As ovarian Sertoli-Leydig cell tumors are extremely rare, it appears worthwhile to report cases with uncommon clinical and/or laboratory findings. A 37-year-old nulligravida female was referred because of secondary amenorrhea and hirsutism for 11 years. She appeared virilized with a male body habitus. The highest plasma testosterone (T) level was 1,070 ng/dl. A laparotomy with salpingo-oophorectomy was performed and the pathological report confirmed the presence of Sertoli-Leydig cell tumor of intermediate differentiation. Ovarian venous T value was 5,690 ng/dl. The patient was alive and well with improved hirsutism 4 years after the operation. The duration of symptoms and T level in our patient were among the highest values ever recorded in the literature. This supports the slowly progressive nature of the tumor and emphasizes that Sertoli-Leydig cell tumors should be included in the differential diagnosis of patients with elevated T level regardless of the duration of clinical symptoms.