Abstract
Introduction: The orthokeratinized odontogenic cyst (OOC) is an uncommon developmental cyst that occurs between the second
to fifth decades, more commonly in males. It is a solitary lesion that mostly occurs in the mandible rather than the maxilla.
Histologic features include a thin, uniform epithelial lining with orthokeratinization and a prominent granular layer below a noncorrugated
onion-skin-like surface.
Case Presentation: A 40-year-old man presented with pain and swelling in the left mandibular canine molar area. The panoramic
radiograph revealed a well-defined radiolucency extending from the left mandibular canine to the left mandibular molar, with scalloped
projections between the teeth roots. Microscopic examination showed a cystic lesion lined by an orthokeratinized stratified
squamous epithelium, and a prominent granular layer beneath the cornified layer was seen. The features were those of an OCC.
Conclusions: From the demographic and radiographic perspectives, the features of OOCs can be similar, but more variation can be
found on routine histopathological analyses.