LAUSR

Introduction First described by Wright in 1981, orthokeratinized odontogenic cyst (OOC) represents a developmental condition derived from epithelial dental lamina rests. Radiographically, OOC appears as a unilocular radiolucency associated with an impacted mandibular third molar. Occasional examples of bilateral/multicentric OOCs have been reported in the literature. Here, we present the clinical, radiographic and microscopic features of a patient with bilateral mandibular OOCs. Case report A healthy 19-year-old male presented with asymptomatic unilocular radiolucencies associated with left and right impacted mandibular 3rd molars. Both molars were extracted and the associated lesions enucleated. Microscopic examination of both specimens showed identical microscopic features consisting of cystic cavities lined by orthokeratinized stratified squamous epithelium with hypergranulosis. A diagnosis of bilateral OOCs was rendered. No recurrence was evident after 4-months. Discussion Six cases (including ours) of bilateral/multicentric OOCs have been documented in the English and Spanish literature. An analysis of the published demographics of this condition showed that most bilateral/multicentric OOCs have a striking predilection for young adult males (age range: 19-41 years; mean age: 27.3 years) with only one case presenting in a female. Bilateral OOCs are almost exclusively associated with impacted mandibular third molars. However, one report documented OOCs in all quadrants. The follow-up period for these cases ranged from 4 months-13 years with no recurrence stated. Conclusion Bilateral OOCs are uncommon and appear to have an excellent prognosis with no recurrence expected. However, more reports with long-term follow-up are needed to draw meaningful conclusions about their biological behavior.