A 20-year-old man complained of increasing pain and swelling in the right ankle joint. Radiographs of the ankle demonstrated an ill-defined osteolytic lesion in the talus associated with a small, round bone formation just proximal to the neck of that bone. Histologic examination of the lesion showed osteoblastic osteosarcoma originating in the talus. Below-knee amputation was performed because of the peripheral location of the tumor and the close anatomic confines of the foot. A literature review demonstrated that the calcaneus and metatarsals are favorite sites of this tumor in the foot, with only one case in the talus. A second case of osteosarcoma in the talus seems to have not been previously reported.