Osteosarcomas have been reported as arising in a number of extraosseous primary sites, most commonly in the retroperitoneum and the muscles of the thighs and limb girdles, but also in a variety of other organs. We present a case arising in the penis, which we believe to be only the fifth reported in the literature and the only documented long-term survivor. Careful histopathological analysis and surgical management remain of key importance in the management of all sarcomas arising in soft tissue.