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Infantile Intraosseous Maxillary Hemangioma. BACKGROUND: Primary osseous hemangiomas of the facial skeleton mimic malignancy. Their location in maxillary sinus, especially in infants is extremely rare. CASE CHARACTERISTICS: 1- month-old full term boy with maxillary swelling. OBSERVATIONS: Biopsy from oral route revealed hemangioma showing vascular channels lined by endothelial cells. Patient improved on oral steroids. MESSAGE: Hemangiomas should be considered as one of the differential diagnosis of unilateral maxillary swelling in infants. Steroids may serve as the primary mode of treatment as opposed to tumor excision.