Isolated ventricular ectopic beats (VEB) are a frequent finding in clinical practice. Determining their etiology and risk stratification strategies requires exclusion of non-cardiac causes and structural heart disease. However, with advances in imaging techniques, molecular biology and electrophysiology, the concept of structural heart disease has changed, and it is important to consider the benefits of using these techniques in cases that have not been entirely clarified. The authors describe the case of a patient with frequent isolated VEB, with a find diagnosis of isolated noncompaction of the lefts ventricle following cardiac magnetic resonance imaging. They present an uncommon cause of VEB, which has raised questions regarding its real prevalence. Better diagnostic accuracy is expected with the use of new imaging techniques.