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E.Ü.T.F. Radyasyon Onkolojisi AD, İzmir In the present paper a patient with primary osteosarcoma of the sphenoid bone which recurred twice following surgery and was managed with re-excision, irradiation and chemotherapy is presented due to its extremely rare presentation and atypical course. An 18-year-old female presented with headache and cranial CT scan revealed a mass destroying the left greater wing of the sphenoid bone. The mass was removed totally and diagnosed as osteosarcoma. Prior to the beginning of adjuvant treatment, during the 4th month after operation a new mass at the lateral wall of the left orbit was demonstrated and the patient was diagnosed as local recurrence and received concomittant chemoradiotherapy. During the 11th month after treatment a new mass on the left frontoparietal scalp which developed due to implantation was noticed and wide local excision was performed. Histologic diagnosis was osteosarcoma. A new course of concomittant chemoradiotherapy was administered. The patient is under follow-up for 4 years free from local recurrence and distant metastases. Keywords : osteosarcoma, sphenoid bone, radiotherapy