Maxillary unicystic ameloblastoma: a rare presentation

Unicystic ameloblastoma (UAM), a rare variant of ameloblastoma, is an odontogenic epithelial neoplasm typically appearing in the mandible. We report an extremely rare case of maxillary UAM with an impacted canine and supernumerary tooth. The patient was a woman in her late 30s who presented with a slight expansion of the left anterior maxilla due to a cystic lesion with impacted teeth. Under a clinical diagnosis of dentigerous cyst, the cystic lesion was completely enucleated by extracting the impacted teeth. Based on the clinical features and pathological findings, the final diagnosis was intraluminal UAM in the anterior maxilla. In the present case, despite the rarity of UAM with impacted teeth in the anterior maxilla, it should be considered during differential diagnosis. Careful clinical examination is required for diagnostic accuracy since the clinical findings of tooth-containing ameloblastoma and dentigerous cyst are very similar.