Oral dirofi lariasis is very rare with non-specifi c clinical manifestations. Here, we report the case of a 65-year-old South American woman with a submucosal nodule on her right buccal mucosa. The nodule was slightly tender and painful. Differential diagnoses included mesenchymal (lipoma or fi brolipoma, solitary fi brous tumor, and neurofi broma) or glandular benign tumors (pleomorphic adenoma) with secondary infections. We performed excisional biopsy. A histopathological examination revealed a dense fi brous capsule and a single female fi larial worm showing double uterus appearance, neural plaque, well-developed musculature and intestinal apparatus. Dirofi lariasis was diagnosed, and the patient was followed-up for 12 months without recurrence.