We report a case of Fusarium oxysporum fungemia that was related to the presence of a central venous catheter and was confirmed by quantitative cultures and scanning electron microscopy of the catheter hub, subcutaneous segment and cuff, and tip. Despite progressive neutropenia, the infection responded to antifungal therapy within 1 day after the catheter was removed. The removal of the catheter could have contributed to the patient's early favorable response.