Ventricular premature beats (VPBs) in a structurally normal heart generally are a benign condition. Rarely, however, reversible cardiomyopathy may develop. This study aimed to evaluate the incidence of cardiomyopathy among pediatric patients in a cohort with frequent VPBs and to examine the characteristics of the ventricular ectopic beats as well as therapeutic options. This study reviewed the charts of all pediatric patients between the ages of 1 day and 18 years seen at the University of Kentucky with the diagnosis of VPBs between 2003 and 2007. Frequent VPBs were defined as an ectopy burden of 5% or more in 24 h. Electrocardiograms, Holter monitors, and echocardiograms were reviewed. The review identified 28 patients (17 boys, age 13.3 ± 5.9 years, and 11 girls, age 13 ± 5.2 years) with frequent VPBs. The echocardiograms of four patients (2 boys, 14%) showed cardiomyopathy. Cardiac function normalized in all four patients, with spontaneous resolution of the VPBs (2 patients) or with antiarrhythmic therapy (2 patients). During a follow-up period of 2.7 ± 2.3 years, 32% of the patients without cardiomyopathy showed a marked spontaneous improvement in arrhythmia burden. Most of the patients showed VPBs with a left bundle branch block (LBBB) and inferior axis morphology. The most commonly associated symptoms were chest pain (17.8%) and dizziness and syncope (21.4%). Generally, VPBs in structurally normal hearts are considered benign. Rarely, a reversible cardiomyopathy can develop, requiring therapeutic intervention.