Abstract Background Premature ventricular contractions (PVCs) are commonly observed during pre-participation cardiac screening in elite athletes. There is an ongoing debate about the clinical significance of PVCs in athletes and whether burden, morphology, or both should be used to differentiate benign PVCs from PVCs suggestive of cardiac disease. Case summary A 28-year-old male athlete was evaluated as part of the pre-participation screening programme. He was asymptomatic, without specific cardiac signs and symptoms. A 12-lead electrocardiogram showed bigeminy PVCs with infundibular morphology and left ventricular outflow tract origin. Left ventricular dilatation and systolic dysfunction without valvular lesions was detected on echocardiography. Cardiac magnetic resonance imaging showed biventricular dilatation and dysfunction without evidence of myocardial fibrosis or fatty infiltration. A 48 h Holter monitoring showed 75191 PVCs (35% of total beats). Radiofrequency ablation was performed, and post-ablation assessments showed no PVCs with normalized ventricular function and dimension. Discussion This case demonstrated that a high PVC burden of common morphology does not also represent a benign finding and requires a comprehensive evaluation to rule out any pathological condition. Furthermore, the present case highlights the critical role of pre-participation cardiac evaluation in identifying cardiac disease in asymptomatic athletes.