Metachronous development of malignant Leydig cell tumor

Leydig cell tumor (LCT), a rare testicular tumor, is malignant in only about 10% of the cases. We report the case of a patient with bilateral malignant LCTs that developed metachronously. After undergoing a right inguinal orchiectomy for a malignant LCT at the age of 43 years, the patient was given cisplatin-based chemotherapy for suspected para-aortic lymph node metastasis. Eighteen months after the right orchiectomy, examination of a left testicular biopsy specimen showed a malignant LCT and a left inguinal orchiectomy was performed. Histologically, the initial malignant LCT exhibited a highly pleomorphic appearance with mitotic figures (58/10 HPF), whereas the second malignant LCT showed fewer mitoses (2/10 HPF). The proliferating cell nuclear antigen (PCNA)-labelling index in these tumors also differed (right-sided tumor, 50%; left-sided tumor, 28%). These findings suggest that the malignant LCT in the left testis developed as a second primary rather than as a metastatic tumor. There have been no known similar cases, although three cases of malignant LCT with contralateral metastasis have been reported.