A 26-year-old woman with a cutaneous granuloma and fistula caused by a dental infection was initially mistaken for a tumor. She was under diagnostic examinations at several health care providers for 1.5 years before a final diagnosis and treatment was reached. The fistula originated from a periapical lesion and extended to the facial skin. Differential diagnoses include malignant tumor, bacterial infection, furuncle and a granulomatous-like lesion. When a patient is presented with a fistula or granuloma-like lesion on the cheek, chin or the submandibular region, a dental infection should always be suspected. Relevant imaging of the teeth and jaws will usually reveal the cause. The medical history, the oral and skin findings, biopsy, imaging results and treatment are presented in this article.