Rhinosporidiosis is a chronic granulomatous inflammatory disease of the mucous membranes caused by Rhinosporidium seeberi. The disease is endemic in India, but is very rare in other countries. A review of the literature shows that our patient is the second documented case from Turkey. Involvement and erosion of the anterior wall of the maxillary sinus as in this case is very rare. A 47-year-old-male is presented with the clinicopathological features and transmission of the disease is discussed.