Background: Unicystic ameloblastoma (UA) is a rare variant of ameloblastoma and represents about 6% of all ameloblastomas. Radiologically, it is very similar to odontogenic jaw cysts due to its unilocular radiolucency. Case Report: This report discusses the clinical, radiological, histopathological and surgical findings of mural unicystic ameloblastoma occupying the right maxillary sinus on a background of dentigerous cyst associated with an impacted tooth in a 22-year-old female patient. Conclusion: It is very uncommon to see this lesion in the maxilla since it usually involves the posterior region of the mandible. Differentiation of an UA from a dentigerous cyst is radiographically difficult when UA is associated with an impacted tooth. Among all UA types, UA with mural proliferation has the highest rate of recurrence. Long-term follow-up for recurrence should be considered for patients with UA.
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