A rare recurrent case of hypervascular juvenile ossifying fibroma

Abstract Juvenile ossifying fibromas (JOFs) are rare fibro-osseous lesions. Conservative surgery, such as curettage and enucleation of the tumor, is the widely accepted therapeutic method; however, JOFs frequently recur after surgery. Here we report a case of a recurrent JOF, which had a hypervascular and aggressive phenotype. A 21-year-old woman presented with a painless, enlarged swelling at the left mandible. Clinical and biopsy findings indicated an ossifying fibroma. The initial tumor was treated via conservative curettage and recurred a year thereafter. The recurrent tumor was treated via segmental bone resection and recurred 2.5 years thereafter. With each recurrence, tumor cell proliferation and tumor hypervascularity increased. We describe the clinical and histological features of a twice-recurrent JOF. Our findings provide new insight into the mechanisms underlying recurrence and may facilitate the establishment of novel diagnostic methods for JOF.