Occult symptomatic bilateral pure Leydig cell tumors in a postmenopausal woman: a case report

Abstract

Background

Pure Leydig cell tumors (LCTs) represent 0.1% of ovarian masses. Postmenopausal patients typically present with virilization. Although LCTs can be challenging to locate on conventional imaging, positron emission tomography (PET) has been demonstrated to be effective.

Case

A 64-year-old postmenopausal woman presented with alopecia, facial hirsutism, and clitoromegaly. Laboratory findings included elevated testosterone and androstenedione. Ultrasound, computed tomography, and magnetic resonance imaging showed no adnexal masses. PET did not demonstrate ovarian fludeoxyglucose-avidity. Histopathology after bilateral salpingo-oophorectomy revealed bilateral Leydig cell tumors. Her testosterone normalized 2 weeks postoperatively.

Conclusion

We describe the occult, symptomatic, bilateral ovarian Leydig cell tumors, an occurrence that has not been described in the literature. Virilizing tumors must be considered in patients with evidence of hyperandrogenism, even without pelvic masses on imaging.

摘要

背景:单纯性间质细胞肿瘤(LCTs)占卵巢肿物的0.1%。绝经后患者的典型表现为男性化。尽管LCTs在常规成像中很难定位, 但正电子发射断层扫描(PET)可以有效定位。

病例:一个64岁的绝经后女性表现为脱发, 面部多毛症和阴蒂肿大。实验室检查发现睾酮和雄烯二酮水平升高。超声、计算机断层扫描及磁共振成像均未见附件肿块。PET未显示卵巢氟代脱氧葡萄糖活动性。双侧输卵管卵巢切除术后的组织病理学检查显示双侧间质细胞肿瘤。该患者睾酮水平于术后2周恢复正常。

结论:我们描述了一种隐匿的、有症状的、双侧卵巢间质细胞肿瘤, 这种肿瘤的发生在文献中从未描述过。在有高雄激素血症证据的患者中, 即使在影像学上没有盆腔肿块, 也必须考虑男性化肿瘤。

Keywords:

• Leydig cell tumor
• virilization
• oophorectomy
• ovary
• adrenal gland

Author contributions

S. Ahmed Hussain was the primary author of the manuscript which was edited by Dr. Dubil. The verbiage for the description of the pathology and the pathology slides themselves were provided by Dr. De Luca-Johnson. Dr. Johnston assisted with the description of the diagnostic and surgical decision-making process.

Disclosure statement

The authors have no conflicts of interest to declare. No financial support was received from any sources. The views expressed in the article are those of the authors and do not necessarily reflect the official policy or position of the Department of the Navy, Department of Defense, or the United States Government. The authors are military service members. This work was prepared as part of their official duties. Title 17 U.S.C. 105 provides that ‘Copyright protection under this title is not available for any work of the United States Government.’ Title 17 U.S.C. 101 defines a United States Government work as a work prepared by a military service member or employee of the United States Government as part of that person’s official duties.