Infection with West Nile virus (WNV) is often characterized by a mild febrile illness, but it can progress to meningitis, encephalitis, flaccid paralysis, and respiratory failure. The neuro-ophthalmological manifestations of this disease are uncommonly discussed. This case describes a 49-year-old undomiciled male who developed WNV flaccid paralysis with ophthalmoplegia. His symptoms began with difficulty in walking and progressed over several days to flaccid paralysis and ophthalmoplegia. Cerebrospinal fluid was positive for WNV immunoglobulin M antibodies and electromyography demonstrated acute denervation in several muscle groups. This is an unusual case of neuro-invasive WNV presenting with flaccid paralysis and ophthalmoplegia.