Abstract
BACKGROUND: Ovarian Leydig cell tumors, rare androgen-producing neoplasms, may present with rapidly progressive virilization. Preoperative localization is frequently challenging, particularly when bilateral ovarian abnormalities coexist or radiologic findings are equivocal. To avoid unnecessary bilateral oophorectomy and preserve fertility in reproductive-aged females, accurate localization is crucial. CASE PRESENTATION: We describe the case of a 42-year-old reproductive-aged female who presented with hirsutism, voice deepening, amenorrhea, and biochemical evidence of severe hyperandrogenism. Imaging revealed a solid nodule-like lesion in the right ovary and a large cystic lesion in the left ovary, making the true origin of androgen excess uncertain. To achieve definitive localization, preprocedural computed tomography (CT) angiography-guided ovarian vein sampling (OVS) was performed, which delineated the venous anatomy and facilitated successful right ovarian vein catheterization. Hormonal analysis revealed markedly elevated testosterone levels in the right ovarian vein, confirming unilateral secretion. Laparoscopic right adnexectomy and left ovarian cystectomy were performed. Pathological analysis confirmed a right ovarian Leydig cell tumor and a benign left ovarian cyst. Postoperatively, serum testosterone levels normalized, menstruation resumed, and virilization features gradually improved over 1 year. CONCLUSION: This case underscores the utility of CT angiography-assisted OVS in localizing androgen-producing ovarian tumors when conventional imaging is inconclusive. Functional confirmation of laterality enabled fertility-sparing surgery (FSS) in a reproductive-aged patient and led to optimal clinical outcomes. Adapting adrenal vein sampling (AVS) methodology to OVS represents a valuable diagnostic approach in selected cases of severe hyperandrogenism.