Uncommon presentation of potential medication-related osteonecrosis of the jaw

BACKGROUND: This article presents a patient with potential atypical medication-related osteonecrosis of the jaw and reviews related literatures. CASE PRESENTATION: A 52-year-old male showed pain in the left buccal area and had numbness on the left lower lip area. He received medications having anti-angiogenic effect for 4 years. He did not receive irradiation of the jaw regions. In histological view, most of the adipocytes were destroyed and disappeared in the scanty vascular marrow tissue, resulting in the replacement of the fatty necrosis with variable sized vacuolated empty spaces. In the immunohistochemistry analysis, the infiltrated macrophages into the marrow stromal tissue were strongly positive for lysozymes. These findings demonstrate that the presented osteonecrosis underwent a chronic and persistent granulomatous inflammatory reaction. CONCLUSIONS: We conclude that the present case might have been caused by anti-angiogenic drug abuse, affecting the reduction of the mandibular marrow vascularity and subsequently inducing fatty necrosis and an extensive osteolytic change of the mandible.