An Atypical Case of Nonsyndromic Multiple Odontogenic Keratocysts in the Maxilla and Mandible

Odontogenic keratocysts (OKCs) represent a type of developmental cyst frequently encountered in the jawbones, most often in the posterior region of the mandible. While the occurrence of multiple OKCs is usually linked with nevoid basal cell carcinoma syndrome, a small proportion of cases – around 5% – have been reported in patients without any syndromic association. We report an unusual case of multiple nonsyndromic OKCs occurring simultaneously in the maxilla and mandible of a 32-year-old male. The patient presented with a complaint of swelling and mild discomfort in the lower right jaw. Radiographic examination revealed several radiolucent lesions in both jaws, displaying a combination of unilocular and multilocular appearances. The patient had a previous history of OKCs 12 years back in both jaw locations, which were well treated and no recurrence noted postoperative. Postsurgical enucleation, histopathological examination done which confirmed the lesions to be OKCs. This case highlights an unusual presentation of multiple nonsyndromic OKCs. Long-term follow-up and comprehensive radiographic and histological evaluations are crucial to monitor recurrence and rule out syndromic associations.