Abstract Cutaneous fistula or oro-cutaneous fistula (OCF) or odontogenic lesions are uncommon conditions that can resemble typical dermatological lesions. These conditions are often misdiagnosed, resulting in delayed treatment, which can cause the lesion to become chronic, the fistula to enlarge, and there may be bone loss. Although well-documented, this condition is rare and frequently misdiagnosed and improperly managed, leading to problems such as sinus drainage, inflammation, pain, aesthetic concerns, prolonged use of antibiotics, and patient discomfort. In this case, it is challenging for general practitioners to distinguish the fistula opening from primary skin conditions, often leading to incorrect referrals to dermatology, being a main concern of maxillofacial surgery. This case report describes a 26-year-old female patient with a two-year history of a non-healing, continuously discharging abscess in the lower part of face region, specifically the chin or submental area, accompanied by chronic pain. She had been misdiagnosed and inadequately treated for two years, which contributed to the persistence of her symptoms. After a prolonged period, she was successfully treated through surgical intervention combined with endodontic treatment under our guidance and treatment protocol, with follow-up for more than a year, resulting in a favorable outcome. The purpose of this case report is to highlight the importance of considering this condition in the differential diagnosis when encountering skin lesions in the head and neck region, emphasizing the need for accurate diagnosis and appropriate management.