Odontogenic keratocyst (OKC) has always been a subject of interest for researchers and clinicians, owing to its unique pathogenesis, aggressive clinical behavior and association with syndromes. Majority of OKCs occur in the mandibular posterior region, and sporadic type of cysts have a predilection to occur in males and in patients of older age groups. The present article reports a case of sporadic OKC presenting in a relatively unusual manner, occurring in the maxillary anterior region of a young female patient. Unlike other reported cases that found OKC prior to commencement of orthodontic treatment, herein it had possibly occurred as a complication of the orthodontic treatment. The article also depicts characteristic microscopic features of OKC such as uniform parakeratinized lining with multiple infoldings, basal palisading, areas of interface separation between the lining and capsule. Disrupted lining with severe inflammatory cell infiltrate in the capsule was indicative of secondary infection
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