The odontogenic keratocyst is a developmental cystic lesion of jaw bones. There is an on-going debate about the pathogenesis of this entity. Odontogenic keratocysts are usually seen intraosseously in jaws with a predilection in the mandibular molar ramus regions. Extra-skeletal variants are reported rarely in the gingiva. Extra-skeletal variants occurring peripherally in other soft tissue components of the oral cavity are extremely rare. Even though histogenesis is ambiguous, such presentation may be related to the tumour-like behaviour of odontogenic keratocysts. Here, we present a case of peripheral odontogenic keratocyst in a 62-year male who presented with complaint of a painless lump on the right buccal mucosa. The diagnosis was made on biopsy of the lesion. The lesion was excised completely. Key Words: Buccal mucosa, Pathology, Keratocyst, Odontogenic.
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