We present a rare case of a 77-year-old Italian woman, admitted to the neurology unit with the diagnosis of West Nile virus encephalitis. Twenty days after the onset of the neurological symptoms, new negative T-waves appeared on the ECG in association with serum elevation of myocardial necrosis enzymes and regional cardiac wall motion abnormalities on echocardiography, so that a coronary angiography was performed. The exam showed significant stenosis on the left circumflex artery, treated with percutaneous coronary intervention. In addition, a cardiovascular magnetic resonance was performed for further investigation: the T2-weighted images revealed edema in the anterior wall and mid-wall late gadolinium enhancement, significant findings of acute myocardial inflammation. Because of the recent diagnosis of West Nile virus encephalitis and the high serum level of specific IgM antibody, the clinical presentation was suggestive of West Nile myocarditis.