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Epithelioid angiosarcoma is a rare malignant tumor that arises from the endothelium of the blood vessels. In the head and neck area, most of these lesions affect the scalp and facial soft tissues, and the maxillary sinus and the maxilla are among the rarest locations involved. In this paper, we present a case of angiosarcoma of the left maxillary sinus, with extension into the left maxilla. We review the literature and discuss the differential diagnosis of endothelial neoplasms and the management and the prognosis of this tumor.