Successful catheter ablation of premature ventricular contractions triggering torsade de pointes in a small infant with histiocytoid cardiomyopathy: a case report

Abstract

Background A short-coupled variant of torsade de pointes (ScTdP) is rare and resistant to medical treatment. There has not been a reported catheter ablation (CA) of a short-coupled premature ventricular contraction (PVC) triggering ScTdP in an infant. Case summary A neonate was referred to our hospital on the day of birth for Wolff–Parkinson–White syndrome, repeated episodes of supraventricular tachycardia, and a left ventricular non-compaction. She underwent CA of an accessory pathway at 72 days of age. On the 5th day after ablation, she had recurrent TdP episodes resistant to various antiarrhythmic drugs and received extracorporeal membrane oxygenation at 86 days of age. She underwent CA of PVCs triggering TdP at 122 days of age and a weight of 3.4 kg. Two types of PVCs triggering TdP were successfully ablated, which originated from the right ventricle (RV). Pre-potentials were recorded at the earliest ventricular activation sites of the targeted PVCs. After the ablation, she had no TdP episodes and the cardiac assist device was removed. However, she died of uncontrolled heart failure at 6 months of age. The histological findings were compatible with histiocytoid cardiomyopathy and abnormal cells were distributed throughout both ventricles. At the ablation site, fibrotic transmural lesions were noted in the RV wall. Discussion The PVCs triggering TdP were successfully ablated in a 4-month-old girl with histiocytoid cardiomyopathy. The PVCs were likely caused by triggered activity and associated with abnormal Purkinje cells.

Case report: high-dose carvedilol as a potential key drug for arrhythmias in histiocytoid cardiomyopathy

“… 3 , 4 Generally, arrhythmia control in histiocytoid cardiomyopathy is challenging with no specific therapy. 5 , 6 To our knowledge, this is the first case report of histiocytoid cardiomyopathy with refractory ventricular arrhythmias controlled by oral medication.…”

“… 3 There is no specific treatment for arrhythmias of histiocytoid cardiomyopathy. Cataldo et al 5 and Hirano et al 6 reported the cases of histiocytoid cardiomyopathy treated with radiofrequency ablation but their outcomes were not satisfactory.…”