A Rare Case of Bilateral Benign Androgen-Producing Large Adrenocorical Adenomas

Abstract

ABSTRACT: Objective: Virilizing tumors of the adrenal gland are very rare, accounting for 5 to 6% of all adrenal tumors. Among them, adrenal carcinomas are more common than benign adenomas.Methods: A 42-year-old woman presented with a 10-year history of progressive virilizing symptoms. Baseline tests were consistent with hyperandrogenism of an adrenal source: total testosterone was 6.62 nmol/L (normal range, 0.3 to 3.78 nmol/L), free androgen index was 27.8% (normal range, 0.5 to 7.3%), and dehydroepiandrosterone sulfate was 14.22 mmol/L (normal range, 1.5 to 7.7 mmol/L). An abdominal computed tomography (CT) scan showed bilateral adrenal tumors. Right side measured 6 × 3 cm heterogeneous with Hounsfield units (HU) of 27 and 60% washout within 10 minutes. Left adrenal mass measured 3.5 × 3.5 cm with HU of 30 and 60% washout within 10 minutes. The patient underwent a laparoscopic right adrenalectomy based on the size and imaging phenotype. The pathology revealed a 4-cm, well-circumscribed adrenal adenoma with no pathologic evidence of malignancy. Postoperatively, there was significant improvement of hyperandrogenism, and the patient conceived 3 months later. However, postdelivery she had recurrence of hyperandrogenism clinically and biochemically, and follow-up CT scan imaging showed the left adrenal mass had increased in size to 4.3 × 3.5 cm from 3.5 × 3.5 cm. Patient underwent laparoscopic left adrenalectomy, and pathology revealed a 6-cm adrenal adenoma with no evidence of malignant potential.Results: Postoperatively, she has been maintained on steroid replacement and remains disease-free after 3 years of follow-up.Conclusion: We report a rare case of benign bilateral virilizing large adrenal tumors in a premenopausal woman.Abbreviations: ACTH = adrenocorticotropic hormone CT = computed tomography DHEA = dehydroepiandrosterone DHEA-S = dehydroepiandrosterone sulfate hCG = human chorionic gonadotropin HU = Hounsfield units LH = luteinizing hormone SHBG = sex hormone–binding globulin