The genus Ustilago inluding Pseudozyma species comprises smut fungi are well known as plant parasites and rarely responsible for human disease.Here, we present an unusual case of Pseudozyma aphidis infection which is described as a complication of leukemia.To our knowledge, this is the first report on complication caused by P.aphidis in leukemia patients.The accurate diagnosis combined with effective anti-fungal therapy may prevent complication from invasive fungal infection.