Secondary osteosarcomas of the maxilla/premaxilla are extremely rare. We describe a case of a 24-year-old man with a metastasis of osteosarcoma to the premaxilla. The patient had undergone hip disarticulation followed by adria based chemotherapy for chondroblastic osteosarcoma of the proximal right femur ten years ago. The metastatic work up did not show liver or lung metastasis. The patient underwent premaxillectomy in February 1999. The histopathology revealed a chondroblatic osteosarcoma consistent with metastasis in the clinical setting. The patient was disease free at the end of five months.