Pigmented villonodular synovitis (PVNS) is a rare proliferative disease affecting joint synovium, tendon sheaths, bursae. The usual treatment for PVNS is a surgical excision. If destructive joint lesions have occurred, complete resections must be performed followed by arthrodesis or arthroplasty. We report a case of a pigmented villonodular synovitis involving an ankle joint which was treated by total ankle replacement for recurrence after simple synovectomy.