Cherubism is a rare non-neoplastic hereditary disease related to genetic mutations characterized by bilateral bone enlargement of the jaws in childhood. Documented long-term follow-up of a series of cases is presented. Four familial and 4 sporadic cases of cherubism have been treated and followed for a mean of 18 years (range, 5 to 32 years). Four of the patients were subjected to cosmetic surgical correction justified by extreme size of the jaws, whereas the other 4 underwent just biopsy or ectopic and impacted teeth removal. Fibro-osseous lesions spontaneously regressed at different levels at variable time intervals. Apparent radiographic osseous repair occurred routinely. In adult life, the patients exhibited normal facial appearance, and radiographs showed almost complete involution of the lesions. Long-term follow-up of these patients is desirable to evaluate progression or involution of the lesions. Apparently, surgical intervention is unnecessary unless significant functional, esthetic, or emotional disturbances develop. Cherubism, first described by Jones, 1 is a benign, self-limiting fibro-osseous disorder characterized by bilateral expansion of the mandible, maxilla, or both. The lesions are usually symmetrical and painless. Frequently, cherubism is accompanied by dental arch and dental eruption abnormalities. Extragnathic skeletal involvement is rare. The rounded facies and occasional upward cast of the eyes with exposure of the sclerae below the pupil give the patient an appearance reminiscent of the cherubs portrayed in Renaissance paintings. Affected children appear normal at birth and are mentally normal. Jaw expansion is noticed within the first years of life, becoming progressively larger until puberty. Lesion regression is expected to occur spontaneously by the end of adolescence, resolving by middle age. 1