A case of acute transverse myelitis associated with ECHO-25 virus infection was reported here with a brief review of the literature. The patient was a 42-year-old house wife. Without any antecedent symptoms, weakness and paresthesia of bilateral lower extremities, and sphincter disturbance developed suddenly. Neurological examination revealed paraplegia of both legs, hyperreflexia of lower extremities, bilateral positive Babinski sign and impairment of all sensory modalities below Th4 level. No abnormality was found in myelography and CT myelography. Abnormal intensity area was found in lower cervical spinal cord on MRI. Serum antibody titer to ECHO-25 virus was elevated in the convalescent stage (X4096), and 7 month later from the onset of neurological symptoms, high value was continued (X128). We diagnosed her illness as acute transverse myelitis complicating ECHO-25 virus infection. In the literature, only 3 cases of transverse myelitis following ECHO virus infection (type 2, 5, 19) were found. This case seemed to be the first case of transverse myelitis associated with ECHO-25 virus infection.