Head and neck osteosarcomas are rare, accounting for 10% or less of all osteosarcoma. In head and neck osteosarcoma, distant metastasis leads to poor prognosis and there are few reports on surgical resection for pulmonary metastasis. Herein we report a case of mandibular osteosarcoma successfully treated with two surgical resections of pulmonary metastases. A 67-year-old man underwent a segmental resection for mandibular osteosarcoma. We administered postoperative radiotherapy and chemotherapy due to a positive margin of the surgical specimen. The patient developed local recurrence at 18 months after the initial surgery, and subsequently underwent salvage surgery. Six months later, lung metastasis was detected, and a thoracoscopic partial lobectomy was performed. However, recurrence of the lung tumor was detected 10 months postoperatively, and a thoracoscopic upper lobectomy was performed. Sixty-five months after the second lobectomy, no evidence of recurrence or metastasis has been detected. In head and neck osteosarcomas, surgical resection should be taken into consideration in patients with pulmonary metastases.
