Abstract

Odontogenic keratocyst (OKC) rarely appears in the buccal space. It is aggressive and infiltrative, and simple enucleation results in high recurrence. This case report describes an OKC located in the buccal space recurring twice in a 62-year-old man with a 25 mm diameter cystic lesion in the right pterygomandibular space. The multicystic lesion was enucleated. Subsequently, a recurrence was observed, and the gourd-shaped recurrent lesion was also enucleated. The difficulty in detaching the mass from the scar tissue resulted in the perforation of the cystic wall. Thereafter, a second recurrence was observed, and the tissue surrounding the unicystic recurrent lesion was excised. The histopathological features were consistent with those of OKC. The present case is the 11th reported case of OKC in the buccal space and the first with apparent recurrence. Since complete enucleation in the pterygomandibular space is difficult, excision along with the surrounding tissues could prevent recurrence.