Caseous rhinitis is known to be very and invariably unilateral disease which was characterized by the accumulation in the nasal cavities and sinuses of an extremely offensive cheese like mass. It may occur at any age, but the greatest number of patients are seen in the third and fourth decades of life. Most of the reported cases are French and Italian. Recently the authors experienced a caseous maxillary sinusitis who were 49 years old male, which was completely recovered after performed Caldwell-Luc's operation. We present a case of caseous maxillary sinusitis with the consideration of literature concerning the same disease.