Periorbital soft tissue swelling may result due to primary orbital pathology or from adjacent facio-maxillary or sino-nasal inflammatory causes. Osteomyelitis of maxilla in the pediatric age group is a rare entity in this era of antibiotics. We present an 11-month-old female infant who was brought with peri-orbital selling and purulent nasal discharge. Computed Tomography showed erosions of the walls of maxillary sinus suggestive of osteomyelitis. Culture of sinus scraping showed Staphylococcus aureus growth and the child improved with intravenous cloxacillin therapy. This case is presented due to the rarity of its presentation in this age group and for awareness to consider this entity in children having fever and peri-orbital swelling.