Behcet's disease is a multi-systemic, inflammatory, and chronic disorder characterized by recurrent oral aphthous ulcers, genital ulcers, uveitis, and other systemic organ involvement. Cardiac involvement in Behcet's disease is rare; however, it plays an important role in prognosis and increases mortality. The current researchers hereby have reported a case of Behcet′s disease with dilated cardiomyopathy. A 28-year-old male patient was presented with constitutional symptoms, oral and genital aphthous ulcers, pseudofolliculitis, tachycardia, arthritis, splenomegaly, erythrocyte sedimentation rate and C-reactive protein elevation, and left ventricular systolic dysfunction with left ventricular ejection fraction of 45%. Azathioprine and prednisolone were begun for the patient.