Keratocyst of the buccal mucosa: is it odontogenic?

Odontogenic keratocyst (OKC) of the buccal mucosa, the diagnosis of which is based on subjective histologic evaluation, is a controversial entity of questionable existence. This report describes 2 rare cases of parakeratinized cyst arising from the buccal mucosa. Case 1 was a 60-year-old man with a 3-cm cyst and case 2 was a 16-year-old boy with a microcyst incidentally discovered on histology. Both lesions were essentially identical in histologic appearance and immunophenotype to intraosseous and gingival OKC, but they were clearly different from orthokeratinized odontogenic cysts and buccal mucosal epidermoid cysts. Step sections failed to reveal any kind of odontogenic tissue or skin adnexa in the cyst wall. These microscopic characteristics reflexively lead to the diagnosis of OKC, if the extragingival occurrence in the buccal mucosa cannot be considered. An alternative nonodontogenic origin includes a keratocyst of the skin, ie, an unusual mucosal presentation of cutaneous keratocyst. Because its true nature, either odontogenic or epidermal, cannot be conclusively proven at this time, we propose a more descriptive and noncommittal term, “mucosal keratocyst,” for a particular cyst in a buccal location that is morphologically indistinguishable from OKC.