The relationship between accessory pathway-mediated ventricular preexcitation and left ventricular dyssynchrony-induced dysfunction has been described in patients with Wolff-Parkinson-White (WPW) syndrome in the absence of sustained supraventricular tachycardia (SVT). Supraventricular tachycardia in infants is usually successfully suppressed with antiarrhythmic medications, but catheter ablation has ultimately been required as definitive treatment in medically resistant cases. Catheter ablation has not been described in young infants for dyssynchrony-related dilated cardiomyopathy in the absence of SVT. We describe a case of an infant with WPW who did not have sustained supraventricular tachycardia, but who developed rapid progression of ventricular dysfunction after birth. Preexcitation could not be medically suppressed but was successfully ablated. This was followed by complete resolution of ventricular dysfunction within 2 months.