BACKGROUND: Primary intracranial osteosarcoma is a extremely rare disease entity. We describe a case of primary intracerebral osteosarcoma in an adult brain. CASE DESCRIPTION: A patient who presented with a 1-week history of headaches, and MRI examination was performed. The immunohistochemical diagnosis confirmed primary intracerebral osteosarcoma. The patient was treated with a surgical resection of the tumor. CONCLUSION: Primary osteosarcomas occurring in the brain are extremely rare. The MRI images did not provide a specific pretreatment diagnosis, and the histopathology was the mainstay in establishing the diagnosis.
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