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Primary osteosarcomas of the skull and skull base are rare, comprising <2% of all skull tumors. Primary osteosarcomas of the skull are aggressive neoplasms composed of spindle cells producing osteoid which have poor outcome.A 33-year-old woman was admitted to our hospital with a major complaint of a growing mass on her left frontal region of the skull for 10 months. Prior to the accurate diagnosis, the mass on her skull was considered to be eosinophilic granuloma.Computerized tomogram (CT) scan of skull revealed a lytic lesion causing destruction of left frontal bone with surrounding soft tissue mass. The histological examination of the lesion showed typical features of osteosarcoma.The patient received 3 surgeries and adjuvant chemotherapy and radiotherapy for the frontal bone lesion.At the last follow-up, after 4 years, the patient was free of disease both clinically and on imaging by magnetic resonance imaging (MRI) scan after 4 years.Because osteosarcoma of skull is a rare disease, the early recognition and correct diagnosis are very important for a better prognosis. It is therefore imperative that clinicians recognize osteosarcoma early to make an accurate diagnosis and complete surgical resection followed by combined chemo-radiation is proved to be one of the most optimal treatment regimens.