West Nile Virus (WNV), a flavivirus, was first documented in the United States by the Center for Disease Control in 1999. WNV has been associated with a variety of clinical features from a subclinical febrile illness to a neuroinvasive disease. Rhabdomyolysis, however, has not been a clinically well-described occurrence during WNV illness. We report a case of a 56 year-old African-American male with WNV infection who developed neuroinvasive encephalitis, flaccid paralysis, and rhabdomyolysis. During his illness, creatinine kinase (CK) values peaked at 45,276 U/L. We discuss the temporal relationship of the development of rhabdomyolysis, the course of his paralytic illness, imaging results, and present supporting evidence that rhabdomyolysis was not a sequel of another clinical condition. With the increasing spread of WNV, it is important to recognize rhabdomyolysis and flaccid paralysis as important manifestations of neuroinvasive WNV infection.