Introduction: Primary intraosseous squamous cell carcinoma (PIOSCC) is carcinoma that occurs within jaw bones exclusively and is considered a rare condition.The exact percentage of malignant transformation in the wall of odontogenic cysts is yet unknown, It varies between 0.01% and 0.02%. Although the process of changing into malignancy is not fully understood, long standing chronic inflammation was reported to be an initiating factor for this transformation.Case Report: We hereby report a case of recurrent odontogenic keratocyst, (OKC) related to the anterior mandibular region. Panoramic radiograph revealed a large radiolucent area which is partially ill defined in some parts. Histological examination of biopsy revealed malignant transformation with dysplastic changes seen in the epithelial lining and areas of frank invasion. Diagnosis of squamous cell carcinoma on top of odontogenic keratocyst was given.
Conclusion:The diagnosis of primary intra osseus carcinoma was given depending on the histological pictures which showed prominent dysplastic changes in the epithelial lining of the cyst together with areas of invasion into the adjacent connective tissue capsule. it was of poor outcome.
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