Odontogenic keratocyst exhibiting dysplastic changes: Report of a new case in a 14-year-old and literature review

Abstract

Odontogenic keratocyst (OKC) is known for its aggressive behavior and a high tendency for recurrence. Malignant transformation in OKC has been widely reported, but comprehensive studies investigating the clinicopathological features and management strategies of dysplastic OKCs are limited. Herein, we report a new case of an OKC exhibiting premalignant changes in a 14-year-old patient and review published cases of dysplastic OKCs. The lesion presented as an incidental radiolucency in the anterior mandible. Histopathological evaluation revealed an intense inflammatory infiltrate in the connective tissue and severe epithelial dysplasia with focal areas exhibiting carcinoma in situ. The dysplastic epithelial lining expressed diffuse reactivity to p53 and displayed a Ki-67 proliferation index of more than 20 %. The lesion was excised by enucleation and thorough curettage. Uneventful healing was noted at the 6-month follow-up appointment. A review of the literature revealed that the average age of diagnosis of dysplastic OKC was 32 years. It showed a preference for the mandible; the most common sites were the anterior and left sides. Most lesions were treated with enucleation; recurrence was reported in one patient. Epithelial dysplasia arising in OKCs is not a frequent finding. A vigilant evaluation of the excised lesions, mainly those displaying significant inflammation, with p53 immunohistochemical analysis may help avoid a misdiagnosis. As OKCs can recur decades after initial treatment, patients with dysplastic lesions should be more closely followed up.