Unicystic ameloblastoma (UA) is one of the variants of ameloblastoma. It manifests as unilocular radiolucency in the mandible or maxilla on X-ray scans. In very rare cases, it can appear as a localized periradicular radiolucent area, imitating a periapical lesion. In this article, we present two cases of UA that were initially misdiagnosed as periapical lesions. Subsequently, surgical enucleation was performed and the diagnosis of UA was confirmed histopathologically.