UNUSUAL DENTAL FOLLICULAR HAMARTOMA ASSOCIATED WITH A DENTIGEROUS CYST WITH FOCAL PARAKERATOSIS: A CASE REPORT AND REVIEW OF THE LITERATURE

Abstract

Introduction Dental follicular hamartoma with central odontogenic fibroma-like features is a rare condition that has been reported primarily in black African teenagers and young adults and is characterized by involvement of multiple teeth that either show amelogenesis imperfecta or enamel dysplasia, hypodontia, open-bite malocclusion, and gingival overgrowth. Case Report We report a case of an unusual dental follicular hamartoma associated with a dentigerous cyst in the left mandible of a 23-year-old male who was otherwise healthy. The patient presented to his oral surgeon with pain in the left mandible. Clinical examination revealed soft tissue swelling and suppuration associated with the distal aspect of tooth #18. A panoramic radiograph revealed a 3.3 cm x 2.3 cm unilocular radiolucency associated with impacted tooth #17 that extended from the superior aspect of the crown to the mandibular notch. This lesion had been present at least four years prior when it measured 2.6 cm x 2.0 cm. Tooth #17 was extracted and the bulk of the lesion was curetted. The biopsy revealed a cellular proliferation of spindled fibroblast-like cells in a delicately and densely collagenous matrix. Scattered throughout were clustered basophilic spherical calcifications associated with condensations of spindled cells, rarely associated with odontogenic rests. A dentigerous cyst was also present. Following this, the residual lesion was curetted and revealed only an inflamed dentigerous cyst. The differential diagnoses for this condition include regional odontodysplasia or unusual hyperplastic dental follicle with dystrophic calcifications. Conclusion We review the past and current literature on dental follicular hamartoma. To the best of our knowledge, our case report represents only the 9th documented case of dental follicular hamartoma and the first not to be associated with any dental-related dysplasia and other dental abnormalities.