Nitrogen containing bisphosphonate (N-BP) therapy is used extensively to treat osteoporosis and osteolytic bone lesions. Recently, a special form of osteonecrosis limited to the maxillofacial skeleton has been discovered especially within those patients who are receiving either long-term N-BP therapy alone and/or associated with invasive dental procedure. Bisphosphonates accumulate almost exclusively in maxillofacial skeleton owing to high bone turn over remodeling to maintain the mechanical competence. The pathogenesis and why it commonly appears in maxillofacial skeleton and the fixed treatment strategies remains unknown. The aim of this study was to improve the clinician understanding of N-BPs associated osteonecrosis of maxillofacial skeleton by reviewing the past 10 year literature.