As chemotherapy has become more effective, patterns of relapse have begun to change. We report a patient with T-cell acute lymphoblastic leukemia (ALL) who had an isolated mandibular relapse that was first manifested as dental pain. Eight other recently reported patients with acute leukemia who relapsed in the mandible are reviewed. In four the relapse was isolated to the mandible; all four had ALL. Local control of disease was achieved with radiotherapy, often combined with chemotherapy. However, our patient and two of the other four subsequently relapsed and died of refractory leukemia.