Left ventricular dysfunction due to frequent isolated premature ventricular complexes originating from the right ventricular outflow tract (RVOT) in patients without structural heart disease and in the absence of sustained ventricular tachycardia is a rare and poorly characterized entity. Successful identification and radiofrequency catheter ablation of the focal source of ventricular ectopy is the most effective treatment for these patients, leading to a complete normalization of ventricular dimensions and contractile function. In this article, we report two cases of left ventricular dysfunction resulting from frequent isolated premature ventricular complexes originating from the RVOT. After successful ablation of the ectopic focus, improvement of functional status and left ventricular function was observed in both patients, confirming the initial diagnostic hypothesis of a cardiomyopathy induced by repetitive ventricular ectopic beats from the RVOT.