Maxillary ameloblastoma is a rare odontogenic neoplasm that is histologically benign and originates from epithelial cells present in bone tissue. If excised through conservative surgery, this tumour has a high relapse rate and is locally aggressive. The risk, in particularly extensive forms, that the ameloblastoma will invade extra-maxillary structures such as the orbit, the pterygomaxillary fossa, the infratemporal fossa and the base of the skull, means that surgical treatment is difficult if it is to be oncologically radical while respecting function and aesthetics. Thus, in these cases a complete and in-depth diagnostic work-up and careful planning of surgical treatment are needed: surgery entails an ablative phase with en-bloc resection of the neoformation to margins free of neoplastic infiltration, and a reconstruction phase that, within a short time-frame, will re-establish functionality and provide a good aesthetic result. Our experience in treating 2 cases of maxillary ameloblastoma is reported.