Odontogenic keratocysts in the pediatric population

Objective: To review the characteristics and treatment of odontogenic keratocysts in the pediatric population at our institution in light of a comprehensive literature review of odontogenic keratocysts in the general population in the hope of elucidating clinical, radiological, or pathological factors that would suggest a different therapeutic approach to odontogenic keratocysts in the pediatric as opposed to the adult population. Design: A 19-year retrospective medical chart review of children with mandibular or maxillary masses of odontogenic keratocyst origin. Setting: Two academic tertiary care institutions. Patients: Eleven children had pathologically confirmed odontogenic keratocysts. Age at diagnosis ranged from 8 to 18 years (mean, 13.4 years). Results: A cystic mass with dentition displacement was characteristic clinically and radiographically. Treatment principally consisted of enucleation with or without extraction of teeth. Follow-up ranged from 1 to 8 years. Seven patients remained free of disease. Recurrences or second primary lesions occurred in 4 patients, all of whom had a family history of nevoid basal cell carcinoma syndrome or multiple cysts suggestive of this diagnosis. The maximum 8-year interval between initial treatment and recurrence is noteworthy. Conclusions: The diagnosis of odontogenic keratocyst deserves consideration in children who have a mass of the mandible or maxilla. The clinical behavior of this lesion in its initial occurrence and response to conservative treatment seems to be similar to that reported in adults. Odontogenic keratocysts, especially those that are multiple or recurrent, should alert the clinician to the possible underlying diagnosis of nevoid basal cell carcinoma syndrome.