Odontogenic keratocyst (OKC) is a developmental odontogenic cyst with specific histopathologic features and clinical behavior. Several investigators suggested that OKCs should be considered as benign cystic neoplasms rather than cysts. OKCs are locally aggressive benign tumors with a high recurrence rate. Usually, multiple OKCs are observed in nevoid basal cell carcinoma syndrome (NBCCS) but about 5% of OKC patients have multiple cysts without an accompanying syndrome.

Mutation in the human homolog of the Drosophila segment polarity gene, “patched” ((PTCH) gene) is seen. Multiple recurrent OKCs without associated systemic findings are related to partial expression of the gene. However, few cases of non-syndromic multiple keratocystic odontogenic tumors have been reported in the literature. This case report presents a rare case of 11-year-old boy with multiple OKCs without any syndromic association. patient underwent surgery for multiple and recurrent cysts in the jaws over a period of 5 years. The patient was screened for syndromic diseases and no signes or symptoms of syndrome were foundmouseout="msoCommentHide('_com_1')" onmouseover="msoCommentShow('_anchor_1','_com_1')">[DM1]