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Research Article Open access CC BY 4.0

Ameloblastic Transformation of a Treated Odontogenic Keratocyst: A Rare Case Report

Jaishri Pagare, Vikrant Kasat, Vidya Bhamare, Pooja Ghorpade

International Journal of Research and Reports in Dentistry · pp. 579–586 · Published 23 Dec 2025

10.9734/ijrrd/2025/v8i2268

Abstract

Odontogenic keratocyst (OKC) is a developmental cyst of odontogenic origin that develops from residual dental lamina. It is characterized by aggressive growth behavior and a high tendency for recurrence. Transformation of its epithelial lining into benign or malignant lesions is uncommon. In contrast, ameloblastoma is a true odontogenic tumor derived from enamel organ–like tissue and lacks differentiation sufficient for enamel production. Ameloblastomatous change arising within an OKC is exceedingly rare, and such cases are classified as combined or hybrid odontogenic   lesions. In this report, we describe a rare case involving a 40-year-old female who had undergone surgical treatment for an odontogenic keratocyst in the left posterior mandible four years earlier and subsequently presented with a recurrent swelling at the same location. Provisionally, it was diagnosed as a case of recurrent OKC, but on histopathological examination it was revealed as follicular ameloblastoma. This article highlights importance of accurate diagnosis of such lesions, for careful management and prevention of recurrence.

Odontogenic keratocyst ameloblastoma recurrent OKC hybrid odontogenic lesions nevoid basal cell carcinoma syndrome

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