Abstract: A 6-year-old boy developed symptoms of rapidly progressive paraplegia, associated with bowel and urinary dysfunction, but without sensory loss. Magnetic resonance imaging (MRI) examination showed diffuse swelling of the lower spinal cord on T1-weighted images. Based on the clinical presentation and MRI findings, a diagnosis of acute transverse myelitis was made. The serum titer of neutralizing antibody against Coxsackie virus B5 rose from 1/4 on admission to 1/256 1 month later and Coxsackie virus B5 was isolated from stool samples. This case serves as a reminder that acute transverse myelitis can be a rare clinical manifestation of Coxsackie virus B5 infection.