Characterization and Management of Arrhythmic Events in Young Patients With Brugada Syndrome


Por: Michowitz Y, Milman A, Andorin A, Sarquella-Brugada G, Gonzalez Corcia MC, Gourraud JB, Conte G, Sacher F, Juang JJM, Kim SH, Leshem E, Mabo P, Postema PG, Hochstadt A, Wijeyeratne YD, Denjoy I, Giustetto C, Mizusawa Y, Huang Z, Jespersen CH, Maeda S, Takahashi Y, Kamakura T, Aiba T, Arbelo E, Mazzanti A, Allocca G, Brugada R, Casado-Arroyo R, Champagne J, Priori SG, Veltmann C, Delise P, Corrado D, Brugada-Terradellas J, Kusano KF, Hirao K, Calo L, Takagi M, Tfelt-Hansen J, Yan GX, Gaita F, Leenhardt A, Behr ER, Wilde AAM, Nam GB, Brugada P, Probst V and Belhassen B

Publicada: 16 abr 2019
Categoría: Cardiology and cardiovascular medicine

Resumen:
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. (C) 2019 by the American College of Cardiology Foundation.

Filiaciones:
Michowitz Y:
 Department of Cardiology, Tel Aviv Sourasky Medical Center, Tel Aviv, Israel

 Sackler School of Medicine, Tel Aviv University, Tel Aviv, Israel

Milman A:
 Sackler School of Medicine, Tel Aviv University, Tel Aviv, Israel

 Department of Cardiology, Leviev Heart Institute, The Chaim Sheba Medical Center, Tel Hashomer, Israel

Andorin A:
 L'institut du Thorax, Service de Cardiologie, CHU de Nantes, Nantes, France

 European Reference Network for Rare and Low Prevalence Complex Diseases of the Heart

Sarquella-Brugada G:
 Pediatric Arrhythmias, Electrophysiology and Sudden Death Unit Cardiology, Department Hospital Sant Joan de Déu, Barcelona-Universitat de Barcelona, Barcelona, Spain

Gonzalez Corcia MC:
 Pediatric Electrophysiology, Cliniques Universitaires St. Luc, Brussels, Belgium

Gourraud JB:
 L'institut du Thorax, Service de Cardiologie, CHU de Nantes, Nantes, France

 European Reference Network for Rare and Low Prevalence Complex Diseases of the Heart

Conte G:
 Heart Rhythm Management Centre, UZ-VUB, Brussels, Belgium

Sacher F:
 Hôpital Cardiologique du Haut-Lévêque & Université Bordeaux, LIRYC Institute, Bordeaux, France

Juang JJM:
 Cardiovascular Center and Division of Cardiology, Department of Internal Medicine, National Taiwan University Hospital and National Taiwan University College of Medicine, Taipei, Taiwan

Kim SH:
 Division of Cardiology, Department of Internal Medicine, College of Medicine, The Catholic University of Korea, Seoul, Korea

Leshem E:
 Department of Cardiology, Tel Aviv Sourasky Medical Center, Tel Aviv, Israel

 Beth Israel Deaconess Medical Center, Boston, Massachusetts

Mabo P:
 Cardiology and Vascular Disease Division, Rennes University Health Centre, Rennes, France

Postema PG:
 European Reference Network for Rare and Low Prevalence Complex Diseases of the Heart

 Heart Centre AMC, Department of Clinical and Experimental Cardiology, AMC, University of Amsterdam, Amsterdam, the Netherlands

Hochstadt A:
 Department of Internal Medicine J, Tel-Aviv Medical Center, Tel Aviv, Israel

Wijeyeratne YD:
 European Reference Network for Rare and Low Prevalence Complex Diseases of the Heart

 Cardiovascular Sciences, St. George's University of London and Cardiology Clinical Academic Group St. George's University Hospitals NHS Foundation Trust, London, United Kingdom

Denjoy I:
 European Reference Network for Rare and Low Prevalence Complex Diseases of the Heart

 Service de Cardiologie et CNMR Maladies Cardiaques Héréditaires Rares, Hôpital Bichat, Paris, and Université Paris Diderot, Sorbonne, Paris, France

Giustetto C:
 Division of Cardiology, University of Torino, Department of Medical Sciences, Città della Salute e della Scienza Hospital, Torino, Italy

Mizusawa Y:
 European Reference Network for Rare and Low Prevalence Complex Diseases of the Heart

 Heart Centre AMC, Department of Clinical and Experimental Cardiology, AMC, University of Amsterdam, Amsterdam, the Netherlands

Huang Z:
 Department of Cardiology, the First Affiliated Hospital of Xiamen University, Xiamen, Fujian, China

Jespersen CH:
 European Reference Network for Rare and Low Prevalence Complex Diseases of the Heart

 The Department of Cardiology, The Heart Centre, Copenhagen University Hospital, Rigshospitalet, Copenhagen, Denmark

 Department of Forensic Medicine, Faculty of Medical Sciences, University of Copenhagen, Copenhagen, Denmark

Maeda S:
 Heart Rhythm Center, Tokyo Medical and Dental University, Tokyo, Japan

Takahashi Y:
 Heart Rhythm Center, Tokyo Medical and Dental University, Tokyo, Japan

Kamakura T:
 Division of Arrhythmia and Electrophysiology, Department of Cardiovascular Medicine, National Cerebral and Cardiovascular Center, Osaka, Japan

Aiba T:
 Division of Arrhythmia and Electrophysiology, Department of Cardiovascular Medicine, National Cerebral and Cardiovascular Center, Osaka, Japan

Arbelo E:
 Cardiology Department, Cardiovascular Institute, Hospital Clinic and IDIBAPS, Barcelona, Catalonia, Spain

Mazzanti A:
 European Reference Network for Rare and Low Prevalence Complex Diseases of the Heart

 Molecular Cardiology, Istituti Clinici Scientifici Maugeri IRCCS, Pavia, Italy

Allocca G:
 Division of Cardiology, Hospital of Peschiera del Garda, Veneto, Italy

Brugada R:
 Cardiovascular Genetics Center, Medical Science Department, University of Girona-IDIBGI (CIBERCV) Cardiology Service, Hospital Josep Trueta, Girona, Spain

Casado-Arroyo R:
 Department of Cardiology, Erasme University Hospital, Université Libre de Bruxelles, Brussels, Belgium

Champagne J:
 Quebec Heart and Lung Institute, Quebec City, Quebec, Canada

Priori SG:
 European Reference Network for Rare and Low Prevalence Complex Diseases of the Heart

 Molecular Cardiology, Istituti Clinici Scientifici Maugeri IRCCS, Pavia, Italy

Veltmann C:
 Rhythmology and Electrophysiology, Department of Cardiology, Hannover Medical School, Hannover, Germany

Delise P:
 Division of Cardiology, Hospital of Peschiera del Garda, Veneto, Italy

Corrado D:
 European Reference Network for Rare and Low Prevalence Complex Diseases of the Heart

 Department of Cardiac, Thoracic and Vascular Sciences University of Padova, Padova, Italy

Brugada-Terradellas J:
 Cardiology Department, Cardiovascular Institute, Hospital Clinic and IDIBAPS, Barcelona, Catalonia, Spain

Kusano KF:
 Division of Arrhythmia and Electrophysiology, Department of Cardiovascular Medicine, National Cerebral and Cardiovascular Center, Osaka, Japan

Hirao K:
 Heart Rhythm Center, Tokyo Medical and Dental University, Tokyo, Japan

Calo L:
 Division of Cardiology, Policlinico Casilino, Roma, Italy

Takagi M:
 Division of Cardiac Arrhythmia, Kansai Medical University Medical Center, Moriguchi, Japan

Tfelt-Hansen J:
 European Reference Network for Rare and Low Prevalence Complex Diseases of the Heart

 The Department of Cardiology, The Heart Centre, Copenhagen University Hospital, Rigshospitalet, Copenhagen, Denmark

 Department of Forensic Medicine, Faculty of Medical Sciences, University of Copenhagen, Copenhagen, Denmark

Yan GX:
 Lankenau Medical Center, Wynnewood, Pennsylvania

Gaita F:
 Division of Cardiology, University of Torino, Department of Medical Sciences, Città della Salute e della Scienza Hospital, Torino, Italy

Leenhardt A:
 European Reference Network for Rare and Low Prevalence Complex Diseases of the Heart

 Service de Cardiologie et CNMR Maladies Cardiaques Héréditaires Rares, Hôpital Bichat, Paris, and Université Paris Diderot, Sorbonne, Paris, France

Behr ER:
 European Reference Network for Rare and Low Prevalence Complex Diseases of the Heart

 Cardiovascular Sciences, St. George's University of London and Cardiology Clinical Academic Group St. George's University Hospitals NHS Foundation Trust, London, United Kingdom

Wilde AAM:
 European Reference Network for Rare and Low Prevalence Complex Diseases of the Heart

 Heart Centre AMC, Department of Clinical and Experimental Cardiology, AMC, University of Amsterdam, Amsterdam, the Netherlands

Nam GB:
 Division of Cardiology, Department of Internal Medicine, Asan Medical Center, University of Ulsan College of Medicine, Seoul, Korea

Brugada P:
 European Reference Network for Rare and Low Prevalence Complex Diseases of the Heart

 Heart Rhythm Management Centre, UZ-VUB, Brussels, Belgium

Probst V:
 L'institut du Thorax, Service de Cardiologie, CHU de Nantes, Nantes, France

 European Reference Network for Rare and Low Prevalence Complex Diseases of the Heart

Belhassen B:
 Department of Cardiology, Tel Aviv Sourasky Medical Center, Tel Aviv, Israel

 Sackler School of Medicine, Tel Aviv University, Tel Aviv, Israel
ISSN: 07351097





JACC-Journal of the American College of Cardiology
Editorial
ELSEVIER SCIENCE INC, STE 800, 230 PARK AVE, NEW YORK, NY 10169, Estados Unidos America
Tipo de documento: Article
Volumen: 73 Número: 14
Páginas: 1756-1765
WOS Id: 000463866300003
ID de PubMed: 30975291
imagen Green Submitted, Bronze

MÉTRICAS