Discussion:
This is the first case reporting the association of Lamin A/C with ventricular noncompaction and a vertical vein connection from the LSPV to the SVC. The occurrence of atrial and ventricular arrhythmias and AV block has been typically observed (3-5). Interestingly in these 2 sisters the atrial arrhythmias arose from the basal interatrial septum, closed to the area where the AV block was present. In addition significant interatrial conduction delay was present. Hence the MRI in one patient did not show any late enhancement nor scar. The disease is inherited as autosomal dominant trait with high penetrance; most importantly it may be associated with an adverse cardiac outcome including heart transplantation and malignant ventricular arrhythmia (5, 6). It is important for the electrophysiologist to diagnose Lamin A/C mutations, as the prognosis is worse and therefore ICD implantation recommended in many cases(7).
Irregular atrial flutters with intraatrial conduction delay are impossible to map with contact mapping systems using summation of serial activation measurements annotated to a reference, because atrial conduction times are not constant. Instead beat to beat analysis of the simultaneous activation in the entire heart chamber shows the origin of arrhythmias at the instant without annotation. Noncontact mapping of the electrical potential has limitations set by the physics of the electrical field (1). But the development of a novel mapping system analyzing the source of the electrical field (charge density and dipole density) has allowed successful treatment of these 2 patients without major arrhythmias over a follow up of 1.5 and 3 years, as demonstrated in PM and ICD interrogation.
Key Teaching Point :
We describe several cardiac disorders associated with Lamin A mutation in the same family including, dilated cardiomyopathy, left ventricular non compaction, complex atrial and ventricular arrhythmias and a vertical vein connection.
Ablation of complex atrial arrhythmias was possible by the use of noncontact dipole density mapping and radiofrequency ablation