A 37-year-old asthmatic presented with arthralgia, dyspnea, hemoptysis, wheezing, abdominal pain, and eosinophilia. He did not receive any corticosteroid therapy previously and five weeks prior to admission Montelukast was started. Transthoracic echocardiography showed severely depressed left ventricular function with an ejection fraction of 20-25%. By the diagnosis of Montelukast-assoclated Churg- Strauss syndrome with cardiac involvement, the drug was stopped and steroids started. During the follow up period of 3 years, he did not have an other vasculitic episode. Although this is a rare association, the clinicians need to be vigilant in all patients who develop systemic symptoms when starting treatment with leukotriene antagonists.