Plexiform ameloblastoma is one of the variant of ameloblastoma, with specific histopathological features. It manifests as unilocular or multilocular radiolucencies in the mandible or maxilla. In very rare cases, it can appear as a localized periradicular radiolucent area and imitate an endodontic lesion. Reported here is a rare case of plexiform ameloblastoma in an uncommon location, which was misdiagnosed as periapical lesion of inflammatory origin and treated endodontically. Surgical enucleation was followed and diagnosis of plexiform ameloblastoma was confirmed on histological grounds.