Leydig-Sertoli cell tumors are rare, with only few cases reported in the literature. We herein present a 41-year-old male who underwent a right orchiectomy for a testicular tumor at the age of 34. Seven years later, he presented at our institution with a large retroperitoneal mass encasing the abdominal great vessels. The patient underwent en-bloc resection of the mass and concomitant infrarrenal aorta and inferior vena cava reconstruction. Pathology report revealed a Leydig-Sertoli cell sex cord-stromal tumor. The patient recovered well from the surgery and he was discharged home in stable condition. To our knowledge this is the first report of a Leydig-Sertoli cell sex cord-stromal tumor with late retroperitoneal metastasis treated by en-bloc resection and vascular reconstruction of the abdominal great vessels.

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