Syndromic Multiple Odontogenic Keratocysts Associated With Impacted Teeth: A Rare Case Report.

INTRODUCTION: Odontogenic keratocysts are developmental odontogenic cysts that may occur as solitary lesions or as multiple jaw lesions. Multiple odontogenic keratocysts in young patients should raise suspicion of an underlying syndrome, particularly Gorlin-Goltz syndrome. CASE PRESENTATION: A 15-year-old male was referred to the Dental Hospital of Hasanuddin University after a 9-month history of progressive left mandibular enlargement. Clinical and radiographic assessment revealed multiple cystic lesions involving the maxilla and mandible, with impacted teeth in several regions. Chest radiography showed bifid left ribs, and computed tomography showed bilateral calcification of the falx cerebri. Histopathological examination confirmed odontogenic keratocystic lesions. THERAPEUTIC INTERVENTION AND OUTCOMES: The patient underwent staged enucleation of multiple maxillary and mandibular cysts under general anesthesia. The postoperative course was uneventful. Obturator rehabilitation was provided, and follow-up radiographic evaluation showed early bone formation. The final diagnosis was syndromic multiple odontogenic keratocysts of the maxilla and mandible associated with Gorlin-Goltz syndrome. CONCLUSION: Multiple odontogenic keratocysts in adolescents require careful syndromic evaluation. Early diagnosis, staged surgical management, long-term surveillance, and multidisciplinary collaboration are essential to reduce recurrence and identify associated systemic manifestations.