We describe the case of a neonate with bacteraemia from whom the recently described organism Arcobacter butzleri was isolated. This appears to be the first report of the organism causing neonatal infection. Clinical details suggest that the infection was contracted in utero, although the mother showed no evidence of disease before delivery. Treatment of the preterm infant was ultimately successful in resolving the infection but the organism proved resistant to a wide range of antibiotics. Similar patterns of antibiotic resistance were also observed in 39 reference and field strains of the genus Arcobacter. These findings, combined with available data on the distribution of Arcobacter species, suggest that these organisms may be important human pathogens. Optimized methods for their isolation and identification are therefore required so as to ascertain their role in human disease.