Context:In West Nile virus (WNV) encephalitis, polio-like illness has been reported but there is no report on acute transverse myelopathy. Design, Setting and Participants:We report a patient with WNV myelopathy admitted in a tertiary care teaching hospital, India along with review of the literature. Findings:A 34 year-old lady presented with fever, headache, diarrhea, seizure, bulbar weakness and quadriplegia for 20 days. Her encephalopathy, bulbar and upper limb weakness improved within few days but flaccid areflexic paraplegia persisted till 6 months with a horizontal sensory level at D3. Electromyography was suggestive of anterior horn cell involvement and somatosensory evoked potential was unrecordable. MRI revealed middle cerebellar peduncle, pons and whole of spinal cord involvement. We could get 11 articles with spinal cord involvement in WNV infection in the medical literature through PubMed search. Their clinical, MRI and electro-diagnostic findings and outcome have been discussed.