OBJECTIVE: The goal of this study was to describe the incidence, treatment, and outcomes of osteosarcomas in the head and neck over 26 years in the province of Alberta, Canada. DESIGN: Retrospective analysis of 14 patients identified and treated in Alberta between 1974 and 1999. Overall and disease-specific survival were the most important outcomes of interest. RESULTS: The average age was 43 years. The maxilla was the most common primary site. The majority of the patients received surgical treatment in combination with adjuvant chemotherapy. Disease-specific survival was 42% at 2 years and 30.1% at 5 years. Four patients developed osteosarcomas in the field of previous radiation. CONCLUSIONS: Osteosarcoma of the head and neck is very aggressive. In our series, mostly high-grade malignancy affected a middle-aged population. Surgical resection is difficult owing to anatomic restraints, and adjuvant chemotherapy should be considered in most patients. The series is notable for the poor outcome, with a 5-year actuarial survival of 30%. This may reflect the lack of a standardized treatment protocol.