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Characterization and Management of Arrhythmic Events in Young Patients With Brugada Syndrome
Yoav Michowitz1, Anat Milman2, Antoine Andorin3
1Department of Cardiology, Tel Aviv Sourasky Medical Center, Tel Aviv, Israel; Sackler School of Medicine, Tel Aviv University, Tel Aviv, Israel.
Insights
Young patients with Brugada syndrome (BrS) experience frequent arrhythmic events (AEs), with high recurrence rates even after treatment. Further research into alternative therapies beyond defibrillators is crucial for managing this high-risk group.
Area of Science:
- Cardiology
- Genetics
- Pediatrics
Background:
- Limited information exists on young Brugada syndrome (BrS) patients experiencing arrhythmic events (AEs).
- Understanding characteristics and risk factors for AEs in this population is critical.
Purpose of the Study:
- To describe the characteristics and management of pediatric and adolescent BrS patients with AEs.
- To identify risk factors associated with recurrent AEs in young BrS patients.
Main Methods:
- A cohort of 57 BrS patients (age ≤20 years) with AEs was analyzed.
- Patients were categorized into pediatric (≤12 years) and adolescent (13-20 years) groups.
- Clinical data, ECG findings, genetic mutations, and follow-up outcomes were assessed.
Main Results:
- The study included 57 young BrS patients with AEs, predominantly males presenting with aborted cardiac arrest.
- Fever-related AEs, spontaneous type 1 ECG, and SCN5A mutations were common.
- High recurrence rates of AEs were observed in both pediatric (68%) and adolescent (64%) groups, with specific risk factors identified for each subgroup.
Conclusions:
- Young BrS patients with AEs constitute a highly arrhythmogenic group.
- Current management strategies are linked to significant AE recurrence.
- Alternative therapeutic approaches beyond defibrillator implantation warrant consideration.
Background:
Information on young patients with Brugada syndrome (BrS) and arrhythmic events (AEs) is limited.
Objectives:
The purpose of this study was to describe their characteristics and management as well as risk factors for AE recurrence.
Methods:
A total of 57 patients (age ≤20 years), all with BrS and AEs, were divided into pediatric (age ≤12 years; n = 26) and adolescents (age 13 to 20 years; n = 31).
Results:
Patients' median age at time of first AE was 14 years, with a majority of males (74%), Caucasians (70%), and probands (79%) who presented as aborted cardiac arrest (84%). A significant proportion of patients (28%) exhibited fever-related AE. Family history of sudden cardiac death (SCD), prior syncope, spontaneous type 1 Brugada electrocardiogram (ECG), inducible ventricular fibrillation at electrophysiological study, and SCN5A mutations were present in 26%, 49%, 65%, 28%, and 58% of patients, respectively. The pediatric group differed from the adolescents, with a greater proportion of females, Caucasians, fever-related AEs, and spontaneous type-1 ECG. During follow-up, 68% of pediatric and 64% of adolescents had recurrent AE, with median time of 9.9 and 27.0 months, respectively. Approximately one-third of recurrent AEs occurred on quinidine therapy, and among the pediatric group, 60% of recurrent AEs were fever-related. Risk factors for recurrent AE included sinus node dysfunction, atrial arrhythmias, intraventricular conduction delay, or large S-wave on ECG lead I in the pediatric group and the presence of SCN5A mutation among adolescents.
Conclusions:
Young BrS patients with AE represent a very arrhythmogenic group. Current management after first arrhythmia episode is associated with high recurrence rate. Alternative therapies, besides defibrillator implantation, should be considered.
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