Cherubism is a rare disease characterized by significant loss of medullary bone which is replaced by excessive amounts of fibrous tissue growth within the mandible and maxilla. We present a case of a 4-year-old boy with a rapidly enlarging mandible and maxilla, causing significant change in the facial contour, malocclusion and phonation difficulties. He was treated with aggressive tumor curettage, lateral mandibular cortex osteotomies with medial repositioning. This allowed obliteration of the enlarged medullary space and restoration of the normal mandibular anatomy. At 12 months postoperatively, the patient had significant improvement in facial contour, normal outward appearance, and stable dentition.