At an estimated incidence of 2 cases per million persons per year, osteosarcoma is the most common primary malignant bone tumor in children and adults, excluding hematopoietic intraosseous tumors. Orbital metastases of osteosarcoma are very rare. Only 5 cases of orbital metastasis of osteosarcoma previously reported in the literature. We report the case of a 19-year-old man with known history of osteosarcoma of right distal femur who presented with acute visual loss and progressive protrusion of his left eye. Orbital CT scan and MRI revealed orbital mass eroding orbital walls and intracranial invasion. He underwent superotemporal orbitotomy for debulking of orbital mass. Histopathological examination (HPE) of the specimen was reported as metastatic osteosarcoma with extensive tumor necrosis. Then he underwent adjuvant chemotherapy and palliative radiotherapy. Although orbital metastasis of osteosarcoma is a rare event, it seems it has had an increasing trend recently. so, making efforts to palliate the patient's symptoms by multidisciplinary teamwork and proper interaction among ophthalmologist, orthopedic surgeons and oncologists is necessary.
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