Odontogenic keratocysts located in the buccal mucosa: A description of two cases and review of the literature

Abstract

Odontogenic keratocysts make up 4%–12% of all odontogenic cysts. Most cysts are sporadic but sometimes they arise in the context of basal cell nevus syndrome (Gorlin syndrome). Most odontogenic keratocysts arise in the posterior region of the mandible, but they can occur anywhere in the jaw. In rare instances, they are located peripherally in the gingiva. Even more rare, they are found in the soft tissues of the mouth. There have been a few case reports and small case series of such peripheral odontogenic keratocysts. Some controversy exists as to whether these truly represent a peripheral counterpart of the intraosseous odontogenic keratocysts and if their origin is at all odontogenic. We hereby present two cases of peripheral odontogenic keratocysts, both being located in the soft tissue of the buccal mucosa, and review the literature on peripheral odontogenic keratocysts.

Peripheral odontogenic keratocyst: Report of two new cases and review of the literature

“…In very rare instances OKC can present as an extraosseous soft tissue lesion with no involvement of the adjacent bone. POKC is expected to represent less than 0,5% of all OKCs, and 35 cases have been reported in the indexed English-language literature up to now (Table I) ( 3 - 24 ). Most cases (22 - 63% - including the present ones) affected the gingival/alveolar mucosa, with predilection for the upper buccal region from incisors to premolars ( Table 1 , Table 1 cont.).…”

“…Most cases (22 - 63% - including the present ones) affected the gingival/alveolar mucosa, with predilection for the upper buccal region from incisors to premolars ( Table 1 , Table 1 cont.). Furthermore, POKC shows a slight male prevalence (1.1:1) and affect patients with a mean of 52 years (ranging from 16 to 83 years old); it usually presents as a soft tissue swelling measuring from 3 to 5 mm ( 3 - 24 ). The present cases have some interesting features as case 1 was located in the posterior maxillary gingiva and presented 15 mm in its largest diameter and case 2 affected a 63-year-old female.…”

“…Follow-up is available for 18 reported POKC and shows a low recurrence rate (3 cases – 17%), whereas the remaining 15 cases (83%) did not show any evidence of local recurrence in a mean of 27 months of follow-up ( Table 1 , Table 1 cont.) ( 3 , 6 , 9 - 12 , 14 , 16 - 19 , 22 - 24 ). Case 1 has been followed-up for 48 months with no signs of local recurrence.…”

Mucosal keratocyst of buccal mucosa: A rare entity

“…Nine cases of soft-tissue/peripheral OKCs located in the buccal mucosa have been documented in the literature, out of which only one of the cases was diagnosed in a female patient. [ 5 ] The mean age of occurrence was 49 years with a range of 16–74 years. [ 2 3 5 6 7 8 ]…”

“…[ 1 3 ] The recurrence rate of peripheral OKCs, excluding the gingival cases, is 12.5%. [ 5 ] This is much lower compared to the recurrence rate of intraosseous OKCs which is about 10%–62%. [ 3 ]…”