• Qingkuo Kong, Yang Yu, Tian Tian, Hongguo Zhang, Meiling Sun, Ruizhi Liu, and Yanhong Liu.
• Center for Reproductive Medicine, Center for Prenatal Diagnosis, First Hospital, Jilin University, Changchun, Jilin 130021, PR China.
• Medicine (Baltimore). 2020 Sep 4; 99 (36): e22085.

RationaleTesticular tumors represent 1% to 1.5% of all tumors in men. Those derived from Leydig cells are rare and account for 1% of testicular tumors. Leydig tumor cells can produce steroid hormones such as estrogen, progesterone and testosterone. The amount and type of hormones secreted by these tumors may produce complicated clinical characteristics in these patients.Patient ConcernsHere, we report a patient with azoospermia, a testicular Leydig cell tumor (LCT), and elevated plasma testosterone levels. We describe the diagnostic and therapeutic experience of this case, and our follow-up of the patient's clinical indicators and fertility status.DiagnosisThe patient was diagnosed with azoospermia and a testicular LCT.InterventionsThe patient underwent testicular tumor removal and long-term follow-up.OutcomesAfter 4 months of follow-up, the patient's semen examination index significantly improved and his wife became naturally pregnant. At 4 months of gestation, the fetus was delivered because of a ruptured amniotic cavity. Twenty-six months after tumor removal, the patient's sex hormone levels had completely returned to normal and spermatogenic function had partially recovered, but there was no natural pregnancy with his partner.ConclusionFor LCTs, testis sparing surgery may provide a safe and feasible option to restore spermatogenic function, although longer-term follow-up is required. Drug assistance may be required to maintain spermatogenic function and achieve fertility, and further research is required.

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