Azoospermia, Atrophy, and Asymmetry: Unilateral Sertoli Cell Tumor as a Rare and Overlooked Cause of Male Infertility: A Case Report and Literature Review.

Sertoli cell tumors are uncommon sex-cord stromal tumors making up < 2% of testicular tumors and have been reported to be associated with endocrine dysfunction that could compromise male fertility. Herein, we present a 34-year-old Pakistani male with primary infertility, azoospermia, enlargement of right testis, and contralateral severe testicular atrophy. Hormone analysis showed decreased testosterone level, high follicle-stimulating hormone, and high levels of estradiol with normal levels of serum tumor markers. Ultrasonography and computed tomography revealed a right intratesticular well-defined mass with severe testicular atrophy of the left testis. Right radical orchidectomy was done. Histopathology revealed benign NOS-type Sertoli cell tumor with positive staining for vimentin and calretinin. Non-tumorous testicular parenchyma showed severe impairment in spermatogenesis with Johnsen score 2-3. After the procedure, normalizing hormones were observed but azoospermia persisted. Our report underscores the occurrence of contralateral testicular atrophy with azoospermia due to a unilateral Sertoli cell tumor probably due to chronic estrogen elevation. Evaluation of patients with Sertoli cell tumors for endocrine disorders, fertility assessments, and discussion of fertility preservation measures including onco-testicular sperm extraction needs to be considered.