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Osteosarcoma is a malignant tumor of mesenchymal cells that have the ability to produce osteoid matrix or mineralized bone. They can involve any bone, but in the jaw region they are uncommon and account for about 6% of all cases. These tumors present as painful and progressively growing masses. Often a sudden, non-traumatic fracture is the first significant clinical symptom. This paper reports a clinical case of osteosarcoma in the body of the mandible. A 25-year-old male patient came to the clinic reporting an intra and extra-oral swelling in the left lower jaw, associated with pain and paresthesia, which lasted four months. The patient reported a previous history of mucoepidermoid carcinoma in the left retroauricular region treated with parotidectomy when he was six years old. The diagnostic hypotheses of Odontogenic Myxoma and Osteosarcoma were raised. Thus, an incisional biopsy was performed, which showed histopathological examination compatible with osteosarcoma. After that, it was decided to perform a left segmental pelvemandibulectomy with histopathological study that confirmed the diagnosis. The case report contributes to the knowledge of health professionals about the osteosarcoma of the mandible since many times this diagnostic hypothesis is not raised because it is a rare situation.