A 4-year-old boy with acute lymphoblastic leukemia (ALL) was inserted a central venous catheter into right vena jugularis interna, whereas on BFM-ALL (Protocol 1) therapy. He developed progressive anemia, thrombocytopenia, schistocytes with triangular or crescent shapes, microspherocytes and teardrop cells in the peripheric blood smear, elevated lactate dehydrogenase, and decreased haptoglobin. Schistocytic hemolytic anemia was considered. On chest radiogram, the central venous catheter (CVC) tip was seen beneath the expected location. An echocardiography revealed that CVC was within the right atrium, in contact with tricuspite leaflets. So, CVC was overhauled and a new one was inserted. After the revision, hemolysis ceased; haptoglobin level normalized.