Agranulocytosis is a rare adverse effect associated with prolonged vancomycin therapy, and is potentially serious, especially in end stage renal disease (ESRD) patients. We describe a continuous ambulatory peritoneal dialysis (CAPD) patient that developed vancomycin-induced agranulocytosis during treatment for methicillin-resistant Staphylococcus aureus (MRSA)-associated external cuff infection and pneumonia. The agranulocytosis was rapidly resolved by granulocyte colony-stimulating factor (G-CSF) therapy and by the discontinuation of vancomycin.