Head and neck osteoblastic osteosarcoma is very rare. The main treatment for head and neck osteoblastic osteosarcoma is surgical resection with wide margins, and the benefit of chemical and radiation therapy is not yet clear, but there is still research report is helpful. Diagnosis by X-ray of this disease alone is extremely difficult, and the clinical manifestations and relatively non-specific, so it is easy to make the wrong diagnosis. This case is a middle-aged male who came to our hospital with tooth discomfort in his right jaw with a right lower lip numbness. Clinical diagnosis is hypercementosis over right mandibular first molar area with osteomyelitis. We performed excision, tooth extraction, and debridement under general anesthesia, pathology reports of malignant osteoblastoma osteosarcoma, then arranged for patients undergoing extensive surgery, the right side supraomohyoid neck dissection, and adjuvant chemotherapy and radiation therapy. Good prognosis is noted of this case until now. Due to the rare cases of particularity, it is to put forward and review the literature.