Bacillus cereus infection is rarely associated with actual infection and for this reason single positive blood culture is usually regarded as contamination. However it may cause a number of infections, such catheter-related bloodstream infections. Significant catheter-related bloodstream infections (CRBSI) caused by Bacillus spp. are mainly due to B. cereus and have been predominantly reported in immunocompromised hosts. Catheter removal is generally advised for management of infection. In this report, catheter-related bacteremia caused by B. cereus in a patient with acute lymphoblastic leukemia (ALL) in Istanbul Medical Faculty was presented.