An International Multi-Center Cohort Study on ß-blockers for the Treatment of Symptomatic Children with Catecholaminergic Polymorphic Ventricular Tachycardia.

Fecha de publicación: 01/02/2022 Fecha Ahead of Print: 07/12/2021

Autores de IIS La Fe

Esther Zorio Grima

Autor

Participantes ajenos a IIS La Fe

Peltenburg PJ
Kallas D
Bos JM
Lieve KVV
Franciosi S
Roston TM
Denjoy I
Sorensen KB
Ohno S
Roses-Noguer F
Aiba T
Maltret A
LaPage MJ
Atallah J
Giudicessi JR
Clur SB
Blom NA
Tanck M
Extramiana F
Kato K
Barc J
Borggrefe M
Behr ER
Sarquella-Brugada G
Tfelt-Hansen J
Swan H
Kammeraad JAE
Krahn AD
Davis A
Sacher F
Schwartz PJ
Roberts JD
Skinner JR
van den Berg MP
Kannankeril PJ
Drago F
Robyns T
Haugaa K
Tavacova T
Semsarian C
Till J
Probst V
Brugada R
Shimizu W
Horie M
Leenhardt A
Ackerman MJ
Sanatani S
van der Werf C
Wilde AAM

Grupos

Cardiopatías familiares, muerte súbita y mecanismos de enfermedad (CAFAMUSME)

Investigador Principal

Aitana Braza Boils

Abstract

Background: Symptomatic children with catecholaminergic polymorphic ventricular tachycardia (CPVT) are at risk for recurrent arrhythmic events. Beta-blockers (BBs) decrease this risk, but studies comparing individual BBs in sizeable cohorts are lacking. We aimed to assess the association between risk for arrhythmic events and type of BB in a large cohort of symptomatic children with CPVT. Methods: From two international registries of patients with CPVT, RYR2 variant-carrying symptomatic children (defined as syncope or sudden cardiac arrest prior to BB initiation and age at start of BB therapy <18 years), treated with a BB were included. Cox-regression analyses with time-dependent covariates for BB and potential confounders were used to assess the hazard ratio (HR). The primary outcome was the first occurrence of sudden cardiac death, sudden cardiac arrest, appropriate implantable cardioverter-defibrillator shock, or syncope. The secondary outcome was the first occurrence of any of the primary outcomes except syncope. Results: We included 329 patients (median age at diagnosis 12 [interquartile range, 7-15] years, 35% females). Ninety-nine (30.1%) patients experienced the primary and 74 (22.5%) experienced the secondary outcome during a median follow-up of 6.7 [interquartile range, 2.8-12.5] years. Two-hundred sixteen patients (66.0%) used a non-selective BB (predominantly nadolol [n=140] or propranolol [n=70]) and 111 (33.7%) used a ß1-selective BB (predominantly atenolol [n=51], metoprolol [n=33], or bisoprolol [n=19]) as initial BB. Baseline characteristics did not differ. The HR for both the primary and secondary outcomes were higher for ß1-selective compared with non-selective BBs (HR, 2.04 95% CI, 1.31-3.17; and HR, 1.99; 95% CI, 1.20-3.30, respectively). When assessed separately, the HR for the primary outcome was higher for atenolol (HR, 2.68; 95% CI, 1.44-4.99), bisoprolol (HR, 3.24; 95% CI, 1.47-7.18), and metoprolol (HR, 2.18; 95% CI, 1.08-4.40) compared with nadolol, but did not differ from propranolol. The HR of the secondary outcome was only higher in atenolol compared with nadolol (HR, 2.68; 95% CI, 1.30-5.55). Conclusions: B1-selective BBs were associated with a significantly higher risk for arrhythmic events in symptomatic children with CPVT compared with non-selective BBs, specifically nadolol. Nadolol, or propranolol if nadolol is unavailable, should be the preferred BB for treating symptomatic children with CPVT.

Datos de la publicación

ISSN/ISSNe:: 0009-7322, 1524-4539
Tipo:: Article
Páginas:: 333-344
DOI:: 10.1161/CIRCULATIONAHA.121.056018
Factor de Impacto:: 8,281 SCImago ℠
Cuartil:: Q1 SCImago ℠

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Métricas

Filiaciones

Peltenburg PJ:: Amsterdam UMC, University of Amsterdam, Heart Centre

Department of Clinical and Experimental Cardiology, Amsterdam Cardiovascular Sciences, Amsterdam, the Netherlands
Kallas D:: BC Childrenâs Hospital, Vancouver, Canada
Bos JM:: Division of Heart Rhythm Services and Pediatric Cardiology, Windland Smith Rice Sudden Death Genomics Laboratory, Mayo Clinic, Rochester, MN
Lieve KVV:: Amsterdam UMC, University of Amsterdam, Heart Centre

Department of Clinical and Experimental Cardiology, Amsterdam Cardiovascular Sciences, Amsterdam, the Netherlands
Franciosi S:: BC Childrenâs Hospital, Vancouver, Canada

Department of Pediatrics, University of British Columbia, Vancouver, Canada
Roston TM:: BC Childrenâs Hospital, Vancouver, Canada

Department of Pediatrics, University of British Columbia, Vancouver, Canada

Center for Cardiovascular Innovation, Division of Cardiology, University of British Columbia, Vancouver, Canada
Denjoy I:: Service de Cardiologie et CNMR Maladies Cardiaques Héréditaires Rares, Hôpital Bichat, Université de Paris, Paris, France
Sorensen KB:: Division of Heart Rhythm Services and Pediatric Cardiology, Windland Smith Rice Sudden Death Genomics Laboratory, Mayo Clinic, Rochester, MN
Ohno S:: Department of Cardiovascular Medicine, Shiga University of Medical Science, Otsu, Japan

Department of Bioscience and Genetics, National Cerebral and Cardiovascular Centre, National Cerebral and Cardiovascular Centre, Suita, Japan
Roses-Noguer F:: Department of Cardiology, Royal Brompton Hospital, London, UK
Aiba T:: Department of Cardiovascular Medicine, National Cerebral and Cardiovascular Centre, Suita, Japan
Maltret A:: Service de Cardiologie et CNMR Maladies Cardiaques Héréditaires Rares, Hôpital Bichat, Université de Paris, Paris, France
LaPage MJ:: Department of Pediatrics, Division of Cardiology, University of Michigan, Ann Arbor, MI
Atallah J:: Cardiology, Faculty of Medicine and Dentistry - Pediatrics Dept., Stollery Children's Hospital, Edmonton, Canada
Giudicessi JR:: Division of Heart Rhythm Services and Pediatric Cardiology, Windland Smith Rice Sudden Death Genomics Laboratory, Mayo Clinic, Rochester, MN
Clur SB:: Department of Pediatric Cardiology, Emma Childrenâs Hospital, Amsterdam UMC, University of Amsterdam, Amsterdam, The Netherlands
Blom NA:: Department of Pediatric Cardiology, Emma Childrenâs Hospital, Amsterdam UMC, University of Amsterdam, Amsterdam, The Netherlands

Department of Pediatric Cardiology, Willem-Alexander Childrenâs Hospital, Leiden University Medical Centre, Leiden, The Netherlands
Tanck M:: Amsterdam UMC, University of Amsterdam, Heart Centre

Department of Clinical and Experimental Cardiology, Amsterdam Cardiovascular Sciences, Amsterdam, the Netherlands
Extramiana F:: Service de Cardiologie et CNMR Maladies Cardiaques Héréditaires Rares, Hôpital Bichat, Université de Paris, Paris, France
Kato K:: Department of Cardiovascular Medicine, Shiga University of Medical Science, Otsu, Japan
Barc J:: Université de Nantes, CNRS, INSERM, lâinstitut du thorax, Nantes, France
Borggrefe M:: Department of Medicine, University Medical Center Mannheim, Mannheim, Germany

German Center for Cardiovascular Research (DZHK), Partner Site Heidelberg/Mannheim
Behr ER:: Cardiovascular Clinical Academic Group and Cardiology Research Centre, Molecular and Clinical Sciences Research Institute, St. Georgeâs, University of London, London, UK

St. Georgeâs University Hospitals NHS Foundation Trust, Cranmer Terrace, London, UK
Sarquella-Brugada G:: Pediatric Arrhythmias, Inherited Cardiac Diseases and Sudden Death Unit, Hospital Sant Joan de Déu, Spain

Medical Science Department, School of Medicine, Universitat de Girona, Spain
Tfelt-Hansen J:: Department of Cardiology, Rigshospitalet, Department of Forensic Medicine, Faculty of Medical Sciences, University of Copenhagen, Copenhagen, Denmark
Zorio E:: Department of Cardiology, Hospital Universitario y Politécnico La Fe, Valencia, Spain

Center for Biomedical Network Research on Cardiovascular Diseases (CIBERCV), Madrid, Spain
Swan H:: Heart and Lung Centre, Helsinki University Hospital and Helsinki University, Helsinki, Finland
Kammeraad JAE:: Department of Pediatric Cardiology, Erasmus MC - Sophia, Rotterdam, The Netherlands
Krahn AD:: Center for Cardiovascular Innovation, Division of Cardiology, University of British Columbia, Vancouver, Canada
Davis A:: The Royal Children's Hospital, Melbourne, Australia

Murdoch Childrenâs Research Institute and Department of Paediatrics, Melbourne University, Melbourne, Australia
Sacher F:: LIRYC Institute, Bordeaux University Hospital, Bordeaux University, Bordeaux, France
Schwartz PJ:: Istituto Auxologico Italiano, IRCCS, Center for Cardiac Arrhythmias of Genetic Origin, Milan, Italy
Roberts JD:: Section of Cardiac Electrophysiology, Division of Cardiology, Department of Medicine, Western University, London, Ontario, Canada

Population Health Research Institute, Hamilton Health Sciences, and McMaster University, Hamilton, Ontario, Canada
Skinner JR:: Cardiac Inherited Disease Group New Zealand, Green Lane Paediatric and Congenital Cardiac Services, Starship Childrenâs Hospital, Auckland, New Zealand
van den Berg MP:: Department of Cardiology, University of Groningen, University Medical Centre Groningen, Groningen, The Netherlands
Kannankeril PJ:: Department of Pediatrics, Monroe Carell Jr Childrenâs Hospital at Vanderbilt, Vanderbilt University Medical Centre, Nashville, TN
Drago F:: Pediatric Cardiology and Cardiac Arrhythmias Unit, Department of Pediatric Cardiology and Cardiac Surgery, Bambino GesÃ¹ Children's Hospital, IRCCS, Palidoro-Rome, Italy
Robyns T:: Department of Cardiovascular Diseases, University Hospitals Leuven, Belgium

Department of Cardiovascular Sciences, University of Leuven, Belgium
Haugaa K:: Department of Cardiology, ProCardio Center for Innovation, Oslo University Hospital, Rikshospitalet, Oslo, Norway

University of Oslo, Oslo, Norway
Tavacova T:: Department of Pediatric Cardiology, Childrenâs Heart Centre, Second Faculty of Medicine, Charles University in Prague

Motol University Hospital, Prague, Czech Republic
Semsarian C:: Agnes Ginges Centre for Molecular Cardiology at Centenary Institute, The University of Sydney, Sydney, Australia

Faculty of Medicine and Health, The University of Sydney, Sydney, Australia
Till J:: Department of Cardiology, Royal Brompton Hospital, London, UK
Probst V:: Université de Nantes, CHU Nantes, CNRS, INSERM, lâinstitut du thorax, Nantes, France
Brugada R:: Centro de InvestigaciÃ³n Biomédica en Red de Enfermedades Cardiovasculares (CIBERCV), Madrid, Spain

Cardiovascular Genetics Center, Institut d'InvestigaciÃ³ Biomèdica Girona (IDIBGI), University of Girona, Girona, Spain

Medical Science Department, School of Medicine, University of Girona, Girona, Spain

Cardiology Service, Hospital Josep Trueta, Girona, Spain
Shimizu W:: Department of Cardiovascular Medicine, National Cerebral and Cardiovascular Centre, Suita, Japan

Department of Cardiovascular Medicine, Graduate School of Medicine, Nippon Medical School, Tokyo, Japan
Horie M:: Department of Cardiovascular Medicine, Shiga University of Medical Science, Otsu, Japan
Leenhardt A:: Service de Cardiologie et CNMR Maladies Cardiaques Héréditaires Rares, Hôpital Bichat, Université de Paris, Paris, France
Ackerman MJ:: Division of Heart Rhythm Services and Pediatric Cardiology, Windland Smith Rice Sudden Death Genomics Laboratory, Mayo Clinic, Rochester, MN
Sanatani S:: BC Childrenâs Hospital, Vancouver, Canada

Department of Pediatrics, University of British Columbia, Vancouver, Canada
van der Werf C:: Amsterdam UMC, University of Amsterdam, Heart Centre

Department of Clinical and Experimental Cardiology, Amsterdam Cardiovascular Sciences, Amsterdam, the Netherlands
Wilde AAM:: Amsterdam UMC, University of Amsterdam, Heart Centre

Department of Clinical and Experimental Cardiology, Amsterdam Cardiovascular Sciences, Amsterdam, the Netherlands

Keywords

*atenolol; *child; *death, sudden, cardiac; *metoprolol; *nadolol; *polymorphic catecholergic ventricular tachycardia; *propranolol

Proyectos asociados

RED INVESTIGACION (RECAVA)

RD06/0014/0004 . INSTITUTO DE SALUD CARLOS III; FUNDACION PARA LA INV BIOMEDICA- HOSPITAL GREGORIO MARAÑON; FUNDACIÓN PARA LA INVESTIGACIÓN DEL HOSPITAL UNIVERSITARIO LA FE DE LA COMUNIDAD VALENCIANA . 2006