Prototheca spp exits throughout nature but only infrequently cause infection in humans. Most of the cases of human infection have involved the skin or subcutaneous tissues, but there have been reports of rare cases of protothecosis of the urinary tract, and disseminated disease. The species most commonly isolated is Prototheca wickerhamii. Few cases of peritonitis due to P. wickerhamii in peritoneal dialysis had been reported. We report a successful treatment of Prototheca peritonitis complicating peritoneal dialysis with amphotericin, itraconazole and removal of the catheter.