A 17-year-old white male patient sought dental treatment because of asymptomatic lesions identified in a routine panoramic radiograph. Intraoral and extraoral physical examinations revealed a small expansion of the left body of the mandible and teeth mobility involving elements 35 to 37. A large unilocular radiolucent lesion with well-demarked borders involving teeth 31 to 38 was noticed. On the right side, a unilocular radiolucent lesion with well-demarked borders was identified adjacent to the distal root of tooth 47. The diagnostic hypotheses included keratocysts related to Gorlin syndrome. Excisional and incisional biopsies were performed on the right and left sides of the mandible, respectively. Histopathologic analysis showed cystic capsules lined by orthokeratinized stratified epithelium presenting a flat surface with the connective tissue capsule and no palisading of the basal cells. The left side lesion has been decompressed, and there were no signs of recurrence after a 4-month follow-up period.