Since the first report of aspergillus sinusitis by Zarniko in 1891, the reported cases of aspergillosis of paranasal sinus have been relatively rare. This disease is usually confined to a single paranasal sinus, especially the maxillary sinus. The symptoms of aspergillosis of the paranasal sinus are variously ranged, and should be suspected when the sinusitis is refractory to antibiotics and antral irrigation. The proper treatment of aspergillosis of the paranasal sinus consists of conservative surgical removal of involved tissue and drainage of sinus. The authors have experienced a case of aspergillosis of maxillary sinus and reported with a review of literatures.