This is the first case report of an exceptional maxillary infection due to Scedosporium prolificans. This recently discovered fungus was identified in the sinus. In the literature, it has been observed at different locations. Identification requires careful sample taking for mycology and pathology studies emphasizing the importance in maxillary surgery. This pathogenic fungus is very invasive, particularly in immunodepressed or immunocompromised patients. Therapeutic modalities vary with the patient's immune status.