An extremely rare case of a conception occurring in a 26-year-old patient with a small virilizing Sertoli-Leydig cell tumor (diameter: 0.6 cm), bilateral polycystic ovaries and non-tumorous adrenal hyperandrogenism is presented. Prepregnancy findings included hirsutism, clitoromegaly, secondary amenorrhea, and elevated peripheral plasma testosterone (T; 5.7 ng/ml). Extensive basal steroid screening, dynamic function tests, conventional radiologic procedures, selective glandular vein catheterization, and laparoscopy failed to localize unequivocally the source of androgen excess, but suggested bilateral adrenal involvement. The patient conceived during the diagnostic work-up; peripheral T levels increased to 12.1 ng/ml within the first trimester. An exploratory laparotomy with left adrenalectomy, right adrenal biopsy and left ovarian wedge resection revealed an incompletely removed Sertoli-Leydig cell tumor, but normal adrenal histology. The pregnancy was terminated, a left oophorectomy and right ovarian wedge resection were performed at 14 weeks' gestation. Subsequently, peripheral androgens returned to normal, regular menses resumed, and hirsutism disappeared. Three years later the patient delivered a healthy female infant.
