Recurrent Psammomatoid Juvenile Ossifying Fibroma with Aneurysmal Bone Cyst: An Unusual Case Presentation

Abstract

Juvenile ossifying fibroma (JOF) is a rare, benign, locally aggressive entity of the extragnathic craniofacial bones with a high tendency towards recurrence. Two distinctive microscopic patterns of juvenile ossifying fibroma have been described: a trabecular juvenile ossifying fibroma (TrJOF) and a psammomatoid juvenile ossifying fibroma (PJOF). Psammomatoid variant is predominantly a craniofacial lesion and occurs rarely in the jaws. The pathognomonic histopathologic feature is the presence of spherical ossicles, which are similar to psammoma bodies. Aneurysmal bone cyst exists as a secondary lesion arising from another osseous condition such as fibrous dysplasia, ossifying fibroma and giant cell granuloma. Very few cases of juvenile psammomatoid ossifying fibroma in association with the secondary aneurysmal bone cyst formation have been reported in the literature. Treatment consists of complete surgical removal; the incomplete excision has been associated with a high local recurrence rate. The authors report a case of recurrence of psammomatoid juvenile ossifying fibroma with aneurysmal bone cyst in an 8-year-old boy emphasizing the point that concomitant occurrence of these locally aggressive lesions requires adequate surgical removal and long-term follow-up.