An unusual case of massive osteolysis destroying the left side of the mandible, the maxilla, the orbit, the cranium bones and the upper cervical spine is reported. The evolution of this disease was observed over a period of 4 years. The literature was reviewed, only 39 cases have been found involving the maxillo-facial bones. In our case, attempts at surgery was without success. However, further radiotherapy with 35 Gy controlled the progression of this osteolysis.