With the wide application of antimicrobial drugs,anticancer drugs and hormones and the advancement of invasive treatment,the morbidity and mortality of invasive fungal infections have been increasing year by year,and some rare fungi have been increasingly recognized as clinically pathogenic,which has attracted clinical attention.The genus Acrophialophora contains 24 species,among which Acrophialophora fusispora and Acrophialophora levis have been reported to be clinically pathogenic.Brain abscesses caused by Acrophialophora levis infection are rare,and the optimal clinical treatment is still unclear.In this article,we analyze the clinical manifestations,imaging changes,pathogenetic examination,and diagnostic process of a patient with brain abscess caused by smooth telangiectasia infection admitted to our hospital,improving the awareness of invasive fungi causing infection with brain abscess.