Cherubism is a rare familial disease of childhood that commonly affects the bilateral mandible and maxilla and typically resolves in adulthood. It has been shown to have a male predilection and has been mapped to the SH3 BP2 gene. Only 2 cases of unilateral cherubism have been documented in the literature; in the first case, the contralateral side was eventually affected. Although rare, unilateral cherubism presents a diagnostic dilemma. This case report describes a unique presentation of unilateral cherubism that progressed to affect the contralateral side and describes some of the considerations in the diagnosis and treatment of unilateral benign giant cell lesions of the jaws.
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