Cholesterol granuloma is usually associated with middle ear disease and is very rare in the paranasal sinuses. We report a case of cholesterol granuloma originating in the maxillary sinus of a 52-year-old female. Endoscopic sinus surgery was performed on the left maxillary sinus, and the cholesterol granuloma was successfully removed by the middle meatal antrostomy. Light microscopic examination showed granulomatous tissue with typical cholesterol clefts, multinucleated foreign body giant cells, small areas of hemorrhage, hemosiderin-laden macrophages and plasma cells. We also describe the details of the endoscopic surgical techniques employed in the treatment of this disorder.