Odontogenic Keratocyst in Syndromic and Recurrent Sporadic Forms: Two Case Reports
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24 mars 2026Authors : Ghada Rekik, Marwa Garma, Chaima Khalifa, Afef Slim, Abdellatif Chokri, Hajer Hentati.
Abstract
Background: Odontogenic keratocyst (OKC) is a developmental odontogenic cyst characterized by locally aggressive behavior and a high recurrence rate. Its clinical and radiological similarity to other odontogenic cysts frequently leads to diagnostic errors. Moreover, its association with nevoid basal cell carcinoma syndrome (Gorlin-Goltz syndrome) adds complexity to diagnosis and management.
Case Reports: Two cases of odontogenic keratocyst highlighting distinct diagnostic challenges. The first case concerns a recurrent OKC initially misdiagnosed as a residual cyst and treated without histopathological confirmation, resulting in delayed diagnosis and recurrence. The second case involves a 22-year-old female referred for multiple jaw radiolucencies, in whom the diagnosis of odontogenic keratocyst led to the identification of Gorlin-Goltz syndrome.
Discussion: These cases emphasize the importance of systematic histopathological examination of all jaw cysts, careful evaluation of recurrent or atypical lesions, and consideration of syndromic associations in young patients or in the presence of multiple lesions. Accurate diagnosis, appropriate treatment, and long-term follow-up remain essential for optimal management of odontogenic keratocysts.
Keywords : Odontogenic keratocyst; Residual cyst; Recurrence; Gorlin-Goltz syndrome; Odontogenic cysts.
