Phialemonium species are emerging as fungal opportunistic pathogens of humans; infections caused by these fungi often have a fatal outcome. We report a series of 4 patients undergoing chronic hemodialysis who developed intravascular infection with Phialemonium curvatum. All isolates were of a distinct morphological type but were shown by partial ribosomal sequencing to be closely related to reference isolates of P. curvatum. Two patients in our case series died; both developed overwhelming infection associated with fungemia and endocarditis. Recent literature corroborates our experience that Phialemonium infection presents unique diagnostic challenges and that optimal management, particularly with regard to antifungal therapy, is not known.