Actinomycotic osteomyelitis of the maxilla presenting with oroantral communication is very rare, herein we report the first case of this condition in association with myiasis. A 50-year-old man reported chronic sinusopathy and a non-healing maxillary lesion, with 30 years of evolution, presenting occasional nasal and intraoral purulent discharge, with foul smell, and recurrent episodes of larvae presence. Cone beam computed tomography showed a large hyperdense image inside the left maxillary sinus, with focal areas with soft tissue density, and extensive discontinuity of the maxillary sinus floor, confirming the oroantral fistula. The necrotic tissue curetted during surgery presented hard consistency, and dark greenish color, and was submitted for histopathological analysis. Microscopically, necrotic bone, masses of filamentous bacteria colonies, compatible with actinomycosis, and large rhomboidal structures surrounded by eosinophilic capsule -suggestive of larvae, were observed. The diagnosis of actinomycotic osteomyelitis with presence of structures compatible with larvae was established.