Background: Ameloblastoma is the most common odontogenic tumor arising from the odontogenic epithelium and is known for its distinct aggressive clinical behaviour and characteristic histologic picture. Very few cases of desmoplastic ameloblastoma with prominent osteoplasia (osteoplastic ameloblastoma) exhibiting formation of metaplastic bone trabeculae rimmed by active osteoblasts have been described. Case Presentation: We report an interesting case of desmoplastic ameloblastoma with prominent osteoplasia (osteoplastic ameloblastoma) in a 38-year-old male presented as a recurrence after 14 years in the anterior mandible. Conclusion: A hybrid lesion of desmoplastic ameloblastoma with osteoplastic pattern (osteoplastic ameloblastoma) needs inclusion of new cases to understand their behaviour. Recurrence of lesion after 14 years of initial surgery in our case presents the importance of regular bi-annual follow-up for lifetime.
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