Odontogenic keratocysts (OKCs) are a commonly occurring, benign cystic intraosseous lesion thought to arise from the cell rests of the dental lamina. Most instances of OKC are commonly found in the posterior aspect of the mandible. In rare instances, there have been reported cases of OKCs arising in the peripheral gingiva, but these cases are few and far between. Rarer still are the few documented cases of OKCs arising in other soft tissues of the mouth. There are many divided opinions on the diagnosis of these lesions and whether they are truly odontogenic in origin, given their location when they are enucleated from a soft tissue area. To the best of our knowledge, we present the first reported case of a patient with a peripheral OKC located in the retromolar trigone. We discuss this unique case and review the current literature on peripheral OKCs.
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