Introduction: Craniofacial osteosarcoma is a rare disease accounting for only 1% of all head and neck malignancies. Its clinical features and optimal treatments are not yet clearly established. Methods: We retrospectively analyzed the clinical course of 15 patients with craniofacial osteosarcoma treated in a single institute. Results: Out of 13 patients who initially underwent primary mass resection, disease recurrence was found in five (36%). Patients with positive surgical margins showed higher recurrence and shorter median recurrence-free and overall survival. Of three patients who had secondary operation after local recurrence, two survived for 44.6 and 64.2 months, respectively. One patient who underwent repeated lung metastasectomy had a disease-free survival of 18.4 months from the first recurrence. Conclusion: The current study demonstrates that positive surgical margins are important predictive factors for recurrence and overall survival. Salvage surgery for local recurrence and metastasectomy for systemic recurrence might have a positive impact on survival.
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