Probiotics are used to restore and maintain the healthy intestinal microflora. Although Saccharomyces cerevisiae (SC) is considered as a non-pathogenic yeast, administration of SC as a probiotic is associated with a rare cause of fungemia in immunocompromised patients with central venous catheter insertion. We encountered a case of SC fungemia in a premature infant who presented with respiratory distress syndrome and had undergone central venous catheterization.