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ABSTRACT The aim of this study is to report an unusual case of actinomycosis in the maxilla region of an asymptomatic patient. A 21-year-old white man was referred for the analysis of panoramic radiography and cone beam computed tomography, where it was observed the presence of a hypodense lesion on the left side of the maxilla, which extended from the maxillary left central incisor to the region of the maxillary left second premolar. During intraoral examination, a depression was observed in the hard palate mucosa, as well as a fistula in the alveolar mucosa close to maxillary left central incisor, which had pulp vitality confirmed by thermal tests. A fistulography was performed, with periapical radiography, where it was found that the fistulous path did not originate from the tooth mentioned above. An incisional biopsy was performed for diagnostic purposes. However, given the inconclusive microscopic findings, four months later, a new biopsy was performed. The histopathological examination revealed the presence of a colony of microoganisms with filamentous pattern of radiated rosette, surrounded by polymorphonuclear inflammatory cells. Based on the morphological characteristics, the diagnosis of actinomycosis was established. The treatment was based on antibiotic therapy. Six months after treatment of the infection, no signs of recurrence were observed, and the patient remains in follow-up. Actinomycosis in the maxilla is an uncommon infection with a predilection for males between the ages of 20 and 60, whose treatment is antibiotic therapy associated or not with surgical excision.