Odontogenic Keratocyst of the Intra-oral Soft Tissues: An Illustrative Case Report and Literature Review

Abstract

Background: The odontogenic keratocyst (OKC) is a relatively common odontogenic developmental intra-osseous jaw lesion. Soft tissue/extra-osseous OKCs are a much less frequently reported variant of the condition. Purpose(s): This piece describes the presentation, radiographic, and histopathological features of an intraoral soft tissue OKC of the buccal gingivae. A literature review was conducted to establish the frequency, presentation, and management of soft-tissue/extra-osseous OKCs. Method(s): The authors depict the presentation, surgical management and photomicrography of a soft tissue odontogenic keratocyst (OKC) of the buccal gingivae. A PubMed literature review was conducted using the terms "odontogenic keratocyst" AND "extra-osseous" or "soft tissue". Result(s): Almost one year following excision, there are no clinical or radiographic changes suggestive of recurrence. The adjacent teeth remain vital and asymptomatic. Histopathologically, no features distinguish a soft tissue OKC from a conventional OKC in the case presented. A PubMed literature review revealed that between 1972 and 2023, there were 59 reported cases of OKC occurring in the soft tissues of the oro-facial region, including the muscles of mastication, buccal gingivae and buccal mucosa. Conclusion(s): Extra-osseous OKCs are a rare variant of OKCs. Management through excision appears to be curative, and the variant has no apparent predilection.Copyright © 2025