Unilateral cherubism: a case report and a review of the literature

An 8-year-old female presented to an oral surgeon with a referral from her pediatric dentist for evaluation of a radiolucency of her right posterior mandible. The patient’s parents reported no significant medical history for the patient, and she had no prior surgical history. She was not taking any medications or supplements. Review of the patient’s panoramic radiograph revealed a multilocular radiolucency of the right mandible posterior to an unerupted #31 and into the ascending-ramus. It appeared well-corticated and was composed of a few medium-sized locules. It appeared separate from the nearby developing tooth #31. The remainder of the panoramic radiograph was unremarkable, with appropriate tooth eruption patterns for the patient’s age and no noted tooth development or symmetric radiograph aberration at tooth site #17. On physical examination, the extraoral and intraoral soft tissues were unremarkable. No expansion was obvious and there were no overlying mucosal findings. The histopathologic features of the lesion were consistent with a giant cell lesion of cherubism. Giant cell lesions of the jawbones are common, but at times require clinical information in addition to the histopathology to render a specific diagnosis. In this case, the extensive eosinophilic cuffing of vessels and sparse hemorrhage suggested cherubism as a possible diagnosis. However, the unilateral presentation was considered unusual. The oral surgeon contacted the patient’s family and they were able to confirm a history of cherubism of the patient’s father. After the diagnosis, no further treatment was rendered. Management of cherubism involves watchful waiting as puberty often halts disease progression. In this case, there has been no progression to date. A review of the literature revealed that unilateral cherubism is rare. Only four cases of unilateral cherubism have been reported and of the four, two progressed to bilateral cherubism with continued monitoring.