Orthokeratinized odontogenic cyst is a rare odontogenic cyst which was considered to be a variant of odontogenic keratocyst, but has been recognized as a separate entity due to its varied behavior and histology. Commonly associated with an unerupted tooth in the posterior mandible, it mimics various lesions like dentigerous cyst, odontogenic keratocyst, unicystic ameloblastoma etc. Orthokeratinized odontogenic cyst was thought to be less destructive compared to odontogenic keratocyst. This case series highlights the clinically aggressive nature of Orthokeratinized odontogenic cyst.In this case series six cases of diagnosed Orthokeratinized Odontogenic cyst have been described along with one case of multiple Orthokeratinized odontogenic cysts in a female patient. The clinical, radiographic and histopathologic features have been described in great detail in order to correlate with the previous literature and highlight their clinically aggressive behavior.Orthokeratinized odontogenic cyst can be deceptive and should be diagnosed appropriately to avoid aggressive treatment protocol. Most of these cysts are associated with inflammation which makes it harder to be diagnosed as the epithelium and connective tissue would have undergone structural changes. Differentiating the cystic lesions of the oral cavity is utmost important to devise appropriate treatment suitable for the patient and for assessing the prognosis, recurrence and rehabilitation to improve the quality of life.
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