Background: Leydig cell tumors are the most common non-germ cell gonadal tumors with apparent increased incidence in the last few years. They are usually benign tumors. We report a case of Leydig cell tumor of testis in a patient presenting atypical features.
Case presentation: A 29-year-old Caucasian man, born with right cryptorchidism, corrected without medical treatment before the age of two years, was diagnosed with Leydig cell tumor. Two years after diagnosis was identified moderately elevated estradiol serum level, in the context of a significant overweight, hormonal changes which had maintained after unilateral orchiectomy and after the patient's return to normal weight. Four years after unilateral orchiectomy, elevated value of estradiol persisted and subdiaphragmatic micro lymphadenopathy was observed.
Conclusions: Despite the favorable evolution of the patient four years after unilateral orchiectomy, long-term follow-up is necessary to exclude recurrence or metastasis to the testis. The endocrine profile and imaging investigations need to be repeated periodically. The changes in the hormonal assay and any new aspects on computed tomography scan can be used as a marker of tumor recurrence and require careful screening and the correct therapeutic decisions.