Nosocomial bacteraemia is associated with significant morbidity, mortality, and cost worldwide, and is most commonly caused by coagulase-negative staphylococci (CONS). Establishing the source of CONS bacteraemia is therefore important in the prevention and management of this infection. CONS infections are presumed to originate at the cutaneous sites of central venous catheters (CVCs), a belief that has led to prevention strategies that focus almost exclusively on the skin. However, mucosal colonisation by CONS is well established, suggesting that mucosal sites might be an important source of CONS bacteraemia. We review the published material that evaluates the source(s) of CONS. We included only studies that used a strict definition of CONS bacteraemia, evaluated skin and other potential sources of CONS, and studied the molecular association between CONS blood isolates and their potential sources. Three published reports fulfilled our criteria. In cancer patients with CONS or CONS bacteraemia, most of the colonising strains that had a molecular match with the strain recovered from the blood of the same patient were mucosal isolates; by contrast, no association was seen between CONS blood and skin isolates. Furthermore, in several patient populations evidence was reported of mucosal colonisation by CONS and in several reports experimental and clinical mucosal translocation of CONS with subsequent bacteraemia was documented. Together these data indicate that mucosal sites are an important source of CONS bacteraemia. Clinical strategies for the treatment of patients with a positive blood culture for CONS, the widespread use of antimicrobial-coated CVCs, and maximum barrier protection for CVC insertion should be reassessed, and strategies to decrease mucosal colonisation by CONS should be developed.