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.