Endoscopic sinus surgery has emerged in the last decade as the treatment of choice for chronic sinusitis. We adopted endoscopic endonasal sinus surgery with preservation of the sinus mucosa as primary treatment for a case of maxillary mycosis, based on papers which showed good results in cases of paranasal sinus mycosis treated with this method. Histopathological examination of caseous masses detected in the maxillary sinus at surgery demonstrated Aspergillus. Postoperative irrigation of the affected sinus with an antimycotic drug was performed, and low-doses of a new macroride and antiinflammatory enzyme preparations were administered orally. Though computed tomography still showed thickened sinus mucosa one week after the operation, the appearance of the mucosa was nearly normal about two and a half months later. Endoscopic endonasal sinus surgery was considered useful in the treatment of maxillary mycosis (Aspergillosis) in this case.