A Case of Excessive Unilateral Ovarian Androgen Production

Abstract

The diagnostic goal in severe hyperandrogenemia is to identify the source of excessive androgen production and to exclude the presence of a neoplasm. In cases with strong clinical suspicion of a neoplasm where ultrasound (US) and computerized tomography (CT) scan are normal, selective retrograde venous catheterization is currently recommended for tumor confirmation and localization. In the presented case, dynamic endocrine testing pointed toward androgen producing tumor. Ultrasound, CT scan, and magnetic resonance imaging (MRI) reported normal abdominal and pelvic organs. Retrograde venous catheterization showed a unilateral ovarian peripheral gradient above non-neoplastic range. Histopathology, however, showed the classical picture of the polycystic ovarian syndrome. Case ReportA 28-year-old female presented with a history of primary infertility for five years and increasing hirsutism for the same duration. She had oligoamenorrhea since her menarche at age 17 years of age. On examination, she was morbidly obese (body mass index 34) with severe hirsutism and a receding temporal hairline but no other signs of virilization. There were no cushingoid features. Basic endocrine workup on cycle day three showed evidence of severe hyperandrogenemia and elevated luteinizing hormone (LH) follicle-stimulating hormone (FSH) ratio (Table 1).The preoperative hormone levels are a mean of three samples tested to eliminate errors due to pulsatile secretion. The hormones were analyzed by radioimmunoassay using second antibody for separation (Pantax, Santa Monica, CA, USA) except for LH, FSH, prolactin, thyroid stimulating hormone, and thyroxine which were analyzed by enzyme immunoassay (Amerlite Diagnostics Ltd, Amersham, England).The adrenal steroid production was suppressed by dexamethasone 2 mg orally every six hours for two weeks. Serum testosterone (T) decreased from 44.3 to 24.7 ng/mL, suggesting partial suppression of androgen secretion. Buserelin 1000 μg/day subcutaneously was added for two weeks. Serum estradiol (E2) dropped from104 pg/ml to 25.0 pg/ml during this period. Serum T further dropped from 24.7 to 20.1 ng/mL. The above results raised the suspicion of androgen-producing tumor. Transvaginal US measured the right ovary as 31 mm and the left ovary as 30 mm in greatest diameter. The ovarian texture was normal. Computerized tomography of the abdomen and pelvis (4 mm continuous slices), showed normal adrenal glands in size and texture. There was no evidence of either polycystic ovaries or a circumscribed tumor. Magnetic resonance confirmed the same findings. Selective retrograde venous catheterization under fluoroscopic control was performed. Through a 6F introducing sheath inserted in the femoral vein, samples were obtained from ovarian and adrenal veins and the lower portion of the inferior vena cava; the results are shown in Table 2. On the basis of significantly elevated right ovarian peripheral gradient (OPG), right salpingo-oophorectomy was performed. Both tubes and ovaries looked unremarkable. The ovary was s...