Abstract

We report a case of an 81-year-old female with an asymptomatic, fluctuant, gingival nodule occurring in the area of the left mandibular first premolar and canine. The clinical impression was that of a gingival cyst of the adult. However, microscopic examination revealed features of an odontogenic keratocyst, including a parakeratinized stratified squamous epithelial lining with a palisading basal cell layer and an absence of rete. A review of the literature revealed 12 previously reported cases of peripheral odontogenic keratocyst with an average age of 51 years and a 1.5:1 female:male ratio. Among these cases, there was a striking predilection for the gingiva in the canine-premolar region (n=9), a presentation similar to that of the gingival cyst of the adult. Two cases were associated with superficial erosion of the underlying alveolar bone. However, all reported lesions fulfilled the histopathologic criteria for an odontogenic keratocyst. One case occurred in a patient with nevoid basal cell carcinoma syndrome. Recurrence was seen after 6 months in the present case and after 7 years in 1 of the previously reported cases. There is some controversy in the literature as to whether to consider this lesion a variant of the gingival cyst of the adult or the soft tissue counterpart of the central odontogenic keratocyst; we favor the latter view given the distinct histopathologic features, recurrence potential, and occasional association with nevoid basal cell carcinoma syndrome.

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