Unicystic ameloblastoma (UA) is a benign epithelial odontogenic tumor of the jaws that commonly occurs in 2nd and 3rd decade of life. In fact, this entity is rare in children under 12 years of age. It is characterised as a distinct variant of ameloblastoma, exhibiting a less aggressive behaviour and a lower rate of recurrence than solid conventional ameloblastoma. There are very few reported cases of UA occurring in children below five years of age. The purpose of this case report is to describe a case of UA involving the crown of an unerupted maxillary second premolar in a 3 year old girl. The pathogenesis, clinical appearance, radiographic presentation, histological findings and management of the tumour have also been discussed.
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