Three cases of catheter infection due to Methylobacterium extorquens are reported. Each patient had a history of acute leukemia and was immunocompromised; two had undergone bone marrow transplantation, and the third was receiving consolidation chemotherapy. All three patients survived after removal of the central venous catheter and antibiotic treatment. The clinical features of these cases are compared with those of the 12 previously reported cases of infection due to Methylobacterium species.