Left ventricular outflow tract (LVOT) may be a source of repeated premature ventricular complexes (PVCs). In symptomatic patients, radiofrequency catheter ablation (RFCA) can be effective, either from endocardial or from epicardial sites. A 50-year-old patient, with dilated cardiomyopathy (DCM) and severe left ventricular (LV) dysfunction, left bundle branch block (LBBB), New York Heart Association (NYHA) class IV, received a biventricular implantable cardioverter/defibrillator (ICD) in 2002. Despite drug therapy, PVCs were frequent (21.019/24 h) including prolonged runs, prompting ICD intervention. Premature ventricular complexes showed an inferior axis morphology, with an R/S ratio in V3>1, suggesting an LVOT origin. Despite the cardiac resynchronization therapy (CRT) device, successful RFCA was performed through the anterior venous branch, with a favourable clinical outcome. To our knowledge, this is the first case describing epicardial RFCA of a PVC focus from cardiac veins in the presence of a CRT device.