A 67-year-old female was referred to the oral and maxillofacial radiology service with a chief complaint of pain of the left mandible of 2 months’ duration. Intraoral examination revealed enlargement of the body of the mandible extending from the left retromolar area to approximately the right canine area. The overlying mucosa was normal. The imaging characteristics were of an extensive multilocularneoplasm with numerous curved coarse septa dividing the lesion into multiple cystic spaces. The appearance was most consistent with an ameloblastoma. Histopathologic examination revealed fibrous tissue with multiple cystic spaces, lined by epithelium that varies in thickness and appearance. Most of the cystic spaces were lined by stratified squamous nonkeratinizing epithelium with plaque like thickenings, characteristic of a botryoid odontogenic cyst. Others areas of lining epithelium showed columnar cells, mucous cells and gland-like structures, characteristic of a glandular odontogenic cyst. The lesion was removed by curettage and has been followed closely for the past 2 years with no evidence of recurrence.