Primary leiomyosarcoma of bone is a rare malignant tumor of smooth muscle. We report a case of low-grade subperiosteal primary bone leiomyosarcoma in the tibial diaphysis, which radiologically appeared to be osteoid osteoma. A 35-year-old man presented with a several-year history of a palpable hard nodule in the distal left leg, which had enlarged and become painful over the previous 2 years. Radiographs showed solid periosteal reaction with a well-defined lytic lesion in the posteromedial cortical border of the left tibial diaphysis. Computed tomography demonstrated a small, well-defined lytic lesion, not calcified, in a subperiosteal location, surrounded by solid periosteal bone formation. The lesion was excised en bloc and the histological diagnosis of a low-grade leiomyosarcoma was made. To the best of our knowledge, the surface location of primary bone leiomyosarcoma has not been previously described in the literature.