Aim: To report a case of a non-neoplastic variant of calcifying odontogenic cyst (COC) with the lining epithelium showing ameloblastomatous proliferation and capsule exhibiting features of a cholesterol granuloma. The importance of delineating this histologic variant from unicystic ameloblastoma and the formation of cholesterol granuloma in this variant is discussed. Background: Calcifying odontogenic cyst is a developmental jaw cyst, which presents itself as both the neoplastic and the non-neoplastic forms. The ameloblastomatous variant of COC is often mistaken for unicystic ameloblastoma and treated aggressively. Case report: A 68-year-old female who presented with a cystic enlargement of the posterior mandible on the right side was suggestive of unicystic ameloblastoma based on radiography and initial biopsy report. Microscopic examination of the excision specimen, however, was fitting in favor of calcifying odontogenic cyst with ameloblastomatous proliferation. Conclusion: Identifying the non-neoplastic ameloblastomatous variant of COC from a cystic ameloblastoma is crucial as the treatment of the two lesions vary considerably. Clinical significance: This case emphasizes the need for thorough examination of the entire surgical specimen before arriving at an appropriate diagnosis.
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