Except for their involvement in dental caries, lactobacilli are usually considered to be non-pathogenic, despite occasional reports of serious infections possibly secondary to underlying predisposing conditions like immunodeficiency, malabsorption syndromes, indwelling catheters and malignancy. Here we report a rare case of urinary tract infection (UTI) in neonate due to Lactobacillus species. Lactobacillus causing UTI in our patient is convincing since the Lactobacillus species was isolated in significant quantities from a catheterized urine sample and responded to treatment. The pathogenesis of this infection in our patient is intriguing yet interesting. The fact that she was not breast fed, female gender and a neonate with diminished immune function may have played a role in lactobacillus urinary tract infection. To the best of our knowledge, however, UTI caused by Lactobacillus species in a neonate have not been reported previously.