The orocutaneous fistulous tract of odontogenic origin is often a diagnostic challenge, due to its rare manifestation and absence of dental signs and symptoms. The odontogenic
cutaneous fistula is often misdiagnosed as a superficial skin lesion of non-odontogenic origin delaying the treatment. The diagnosis and treatment must be precise and swift to improve the clinical outcome and minimize the complications. This article presents a rare case of odontogenic
keratocyst involving a linguoverted impacted third molar presenting as orocutaneous
fistula. The patient was initially treated with empirical
antibiotic therapy with no resolution of the
cutaneous fistula and thickening of the skin around the sinus opening resulting in cosmetic
deformity. Once the
fistula was attributed to the underlying
odontogenic cyst, treatment was done by
cyst enucleation along with the extraction of tooth and
fistula excision. The purpose of the paper is to emphasize the importance of early and accurate diagnosis and prompt management of the orocutaneous
fistula due to the odontogenic origin.