Salim Michael, Dasaraju Sandhyarani, Erickson Britt, Khalifa Mahmoud, Burmeister Lynn A
Endocrinology, Diabetes, and Metabolism, University of Minnesota School of Medicine, Minneapolis, USA.
Laboratory Medicine and Pathology, University of Minnesota School of Medicine, Minneapolis, USA.
Cureus. 2024 Mar 1;16(3):e55334. doi: 10.7759/cureus.55334. eCollection 2024 Mar.
Hyperandrogenism in postmenopausal females may arise from either ovarian or adrenal sources and can pose a challenging diagnostic dilemma. We present the case of a 66-year-old female with postmenopausal hyperandrogenism with virilization, adrenal incidentaloma, and concurrent finding of two extremely rare ovarian tumors, including bilateral Leydig cell tumor and Brenner tumor. Laboratory tests showed elevated testosterone and androstenedione and normal dehydroepiandrosterone sulfate (DHEAS). Response to 1 mg overnight dexamethasone suppression test demonstrated persistently elevated testosterone and incomplete suppression of androstenedione. Computed tomography (CT) scan showed a left adrenal nodule and an unremarkable appearance of the ovaries. The pelvic ultrasound did not show an ovarian tumor on the right ovary, and the left ovary was not seen. Adrenal and ovarian vein sampling suggested the ovaries as the source of the testosterone. Given the ovarian vein sampling results, a multidisciplinary discussion between endocrinology and gynecologic oncology concluded that bilateral salpingo-oophorectomy (BSO) was the next best step for diagnosis and management. Laparoscopic BSO was performed. Histopathology showed bilateral Leydig cell tumors and a left ovarian Brenner tumor. At one-year postoperative follow-up, alopecia improved, and testosterone level normalized. This case highlights the importance of diagnostic pathways and interdisciplinary collaboration in managing rare clinical scenarios of hyperandrogenism in postmenopausal females. As in our case, surgeons may be hesitant to remove normal-appearing ovaries. While the three presented tumor types in this case arise from distinct tissues and exhibit different histological characteristics, the presence of such a unique triad prompts consideration of potential unifying pathogenic mechanisms.
绝经后女性的高雄激素血症可能源于卵巢或肾上腺，这可能会带来具有挑战性的诊断难题。我们报告了一例66岁绝经后高雄激素血症伴男性化、肾上腺意外瘤的女性病例，同时发现了两种极为罕见的卵巢肿瘤，包括双侧睾丸间质细胞瘤和勃勒纳瘤。实验室检查显示睾酮和雄烯二酮升高，硫酸脱氢表雄酮（DHEAS）正常。过夜地塞米松抑制试验（1mg）结果显示睾酮持续升高，雄烯二酮抑制不完全。计算机断层扫描（CT）显示左肾上腺结节，卵巢外观无异常。盆腔超声未显示右侧卵巢有肿瘤，左侧卵巢未见。肾上腺和卵巢静脉采血提示卵巢是睾酮的来源。鉴于卵巢静脉采血结果，内分泌科和妇科肿瘤学进行了多学科讨论，得出结论认为双侧输卵管卵巢切除术（BSO）是下一步诊断和治疗的最佳选择。遂进行了腹腔镜BSO手术。组织病理学显示双侧睾丸间质细胞瘤和左侧卵巢勃勒纳瘤。术后一年随访时，脱发改善，睾酮水平恢复正常。该病例强调了诊断途径和跨学科合作在处理绝经后女性高雄激素血症罕见临床情况中的重要性。就像我们的病例一样，外科医生可能会犹豫是否切除外观正常的卵巢。虽然本病例中出现的三种肿瘤类型起源于不同组织，具有不同的组织学特征，但这种独特三联征的存在促使我们考虑潜在的统一致病机制。
