Multiple Odontogenic Keratocysts in a Non-Syndromic Patient: A Case Report and Review of Literature

Odontogenic keratocyst (OKC) is an aggressive cystic lesion that arises from remnants of dental lamina. It mostly affects the mandibular posterior region and is commonly associated with an impacted tooth. Radiologically, it can present as unilocular or multilocular radiolucency with a sclerotic border, causing cortical plate expansion and even sometimes leading to perforation. The occurrence of multiple OKCs is considered the initial presentation of many syndromes. However, cases of multiple OKCs have also been reported in non-syndromic patients. We present a case of 17/M with a complaint of swelling in the mandibular anterior region. CBCT revealed three unilocular expansile lytic lesions in the mandible: a large lesion extending from tooth 46 (mandibular right first molar) to tooth 34 (mandibular left first premolar), along with separate lesions associated with teeth 38 and 48 (mandibular left and right third molar regions). Incisional biopsy was suggestive of OKC. So, cyst enucleation was done under general anaesthesia and histopathology showed a cystic lumen lined by OKC lining. Numerous daughter cysts were also seen. This case emphasizes that the occurrence of multiple OKCs warrants evaluation for possible associated syndromes. Syndromic OKCs typically present at a younger age, show equal involvement of the maxilla and mandible, and demonstrate a higher recurrence rate compared to sporadic cases. Therefore, long-term follow-up is essential to achieve favorable outcomes, and management of such lesions requires a multidisciplinary approach.