True Unilateral Mandibular Cherubism: A Literature Review and Case Report

Cherubism is a self-limiting fibro-osseous disorder that is classically characterized by bilateral mandibular swelling in the first or second decade of life. Three cases of unilateral cherubism have been described in the literature; however, bilateral involvement did not eventually develop in only 1 case. Histopathologic variation and genetic heterogeneity complicate the diagnosis and treatment of a unilateral benign giant cell lesion of the jaws. Our case report presents a 13-year-old male patient with unilateral posterior mandibular swelling that proved to be histologically and clinically consistent with unilateral cherubism. Involution of the unilateral lesion with no bilateral involvement occurred during 8 years of serial examination.