Simultaneous Sertoli Cell-Only Syndrome and Leydig Cell Tumor in a Patient with Azoospermia: A Rare Case Report

Abstract

Testicular cancers comprise 1-1.5% of entire cancers in men, and sex cord-stromal tumors include 5% of testicular cancers. This study aims to report a simultaneous Sertoli cell-only syndrome and Leydig cell tumor in the same patient. A 32-year-old man presented with a history of primary infertility for 3 years. Physical examination revealed normal secondary sexual characteristics. Two successive seminal fluid analyses revealed azoospermia. A scrotal ultrasound scan showed a 28 × 27 mm hypoechoic and hypervascular right testicular mass. Right radical orchiectomy and simultaneous left testicular biopsy were conducted. The histopathological examination revealed Sertoli cell-only syndrome and Leydig cell tumor with focal Leydig cell hyperplasia. Reversing fertility following the management of Leydig cell tumor is rarely mentioned in the literature. A study revealed that fertility recovered following 4 months of management in a primary infertile male. However, infertile men with nonobstructive azoospermia due to SCOS can only have a child by testicular sperm extraction technique. Despite the rare occurrence of Leydig cell tumor, it could be seen in association with Sertoli cell-only syndrome in infertile men with azoospermia. Clinical examination and imaging studies are important in these patients as the possibility of having a testicular mass is high among them.

Keywords: Infertility; Leydig cell tumor; Sertoli cell-only syndrome; Testicular tumor.

Fig. 1.

A U/S scan of the left testis showing hypoechoic and hypervascular mass.

Fig. 2.

Diffuse polygonal cells with abundant eosinophilic cytoplasm (white arrow). Absence of germ cell in the seminiferous tubules with only Sertoli cells (star).

Fig. 3.

Diffuse positive reaction of tumor cells to inhibin stain.