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The orthokeratinized odontogenic cyst (OOC) and odontoma are the odontogenic cyst and tumor respectively that are minimally invasive neoplasms of head and neck region. OOC is a rare variant of odontogenic cyst characterized by the presence of excessive orthokeratin covering the cystic lining. Odontoma is a benign neoplasm/hamartoma often discovered accidently on panoramic radiographs. We came across a case of a 26-year-old male with swelling on his face along with difficulty in breathing. On the basis of radiographic and histopathological findings the final diagnosis of OOC associated with odontoma was given. However, there is no report in the English literature of the simultaneous occurrence of these two lesions and hence this case is very rare. It is unclear whether the two lesions were just coincidental or were actually related to each other.

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