Abstract: Curtobacteria are beaded, filamentous, gram-positive rods found in the environment. Based on detailed phenotypic and chemotaxonomic investigations, there are 7 recognized Curtobacterium species with only one documented report involving their isolation from humans. Furthermore, there have been no previous publications showing definitive identification of a specific Curtobacterium species in blood cultures or other normally sterile body sites. We report a rare and unique case of a brain abscess caused by 2 Curtobacterium species: C citreum and C flaccumfaciens. To the best of our knowledge, this is the first case of Curtobacterium brain abscess reported in the literature.