Furuncular myiasis is a parasitic infection caused by the larvae of Dipteran insects after penetrating healthy skin, causing an erythematous nodule with a central pore. Additionally, the patient may feel a sensation of movement and intermittent pain. Furuncular myiasis affecting the oral and perioral region is rare, usually leading to misdiagnosis. This paper describes a rare case of furuncular myiasis in a 20-year-old healthy male patient who presented an erythematous nodule in the lower lip. An exploratory surgery was indicated for both diagnostic and curative purposes, with successful healing observed in the following appointment. After 3 years of follow-up, the patient is recovered, with no clinical signs or symptoms of the disease.