Abstract

Introduction: Keratocystic odontogenic tumour (KCOT) is a distinct lesion of odontogenic origin. Cases may be sporadic or associated with Gorlin syndrome. Treatment is controversial.

Case report: A case of a 26 year old male with histopathologically proven Keratocystic odontogenic tumour of maxillary sinus with fistulous tract leading from maxillary sinus to infra orbital margin is presented. It was treated with enucleation and fistulectomy. Follow up was uneventful.

Discussion: Keratocystic odontogenic tumour (KCOT) is a benign lesion with aggressive potential. There are many treatment modalities. Present case is being highlighted because of a novel clinical presentation of an associated fistula in absence of antecedent trauma.

Conclusion: Keratocystic odontogenic tumour (KCOT) may have atypical presentation and hence should be considered in differential diagnosis of all maxillary/mandibular cysts.

Keywords: Keratocystic odontogenic tumour (KCOT); Fistula