CaseA case of Leydig cell tumor, associated with azoospermia, is presented.OutcomeThe levels of sex hormones obviously were decreased, including luteinizing hormone (LH) and follicle‐stimulating hormone (FSH), with elevated testosterone. Computed tomography revealed no adrenal gland tumor, but a significant calcification in the right scrotal content was observed. He received a right radical orchiectomy and then he was unable to ejaculate. An endocrine panel revealed significantly decreased levels of testosterone and the low LH level had remained. Hormone replacement therapy with combined LH and FSH successfully recovered and preserved spermatogenesis.ConclusionsAlthough the patient’s sexual function deteriorated after surgery, hormone replacement therapy was successful in establishing spermatogenesis.