Mycobacterium brumae is a rapidly growing environmental mycobacterial species identified in 1993; so far, no infections by this organism have been reported. Here we present a catheter-related M. brumae bloodstream infection in a 54-year-old woman with breast cancer. The patient presented with high fever (39.7 degrees C), and >1,000 colonies of M. brumae grew from a quantitative culture of blood drawn through the catheter. A paired peripheral blood culture was negative, however, suggesting circulational control of the infection. The patient was treated empirically with meropenem and vancomycin, and the fever resolved within 24 h. The catheter was removed a week later, and from the tip M. brumae was isolated a second time, suggesting catheter colonization. The organism was identified by colonial morphology, sequence analysis of the 16S rRNA gene, and biochemical tests.