To improve the dentist′s understanding on odontogenic fistula of skin in the maxillofacial region.

A case of misdiagnosed odontogenic fistula on a patient′s anterior neck skin was retrospectively analysed.

The anterior cervical cutaneous fistula was caused by chronic odontogenic infection, mainly for the corresponding parts′ chronic periapical periodontitis. After the pathogenic tooth extraction and fistula curettage treatment, the cutaneous fistula was healed without recurrence during the follow up.

Clinicians should maintain sharp vigilance on odontogenic infection on the case of maxillofacial cutaneous fistula and determine the etiology to avoid misdiagnosis and mistreatment.