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An international multicenter cohort study on implantable cardioverter-defibrillators for the treatment of symptomatic children with catecholaminergic polymorphic ventricular tachycardia
A. Lamba, TM. Roston, PJ. Peltenburg, D. Kallas, S. Franciosi, KVV. Lieve, PJ. Kannankeril, M. Horie, S. Ohno, R. Brugada, T. Aiba, P. Fischbach, L. Knight, J. Till, SY. Kwok, V. Probst, D. Backhoff, MJ. LaPage, AS. Batra, F. Drago, K. Haugaa,...
Language English Country United States
Document type Journal Article, Multicenter Study
- MeSH
- Defibrillators, Implantable * MeSH
- Child MeSH
- Polymorphic Catecholaminergic Ventricular Tachycardia MeSH
- Tachycardia, Ventricular * therapy physiopathology MeSH
- Humans MeSH
- Adolescent MeSH
- Death, Sudden, Cardiac * prevention & control etiology MeSH
- Follow-Up Studies MeSH
- Child, Preschool MeSH
- Retrospective Studies MeSH
- Ryanodine Receptor Calcium Release Channel genetics MeSH
- Treatment Outcome MeSH
- Check Tag
- Child MeSH
- Humans MeSH
- Adolescent MeSH
- Male MeSH
- Child, Preschool MeSH
- Female MeSH
- Publication type
- Journal Article MeSH
- Multicenter Study MeSH
BACKGROUND: Catecholaminergic polymorphic ventricular tachycardia (CPVT) may cause sudden cardiac death (SCD) despite medical therapy. Therefore, implantable cardioverter-defibrillators (ICDs) are commonly advised. However, there is limited data on the outcomes of ICD use in children. OBJECTIVE: The purpose of this study was to compare the risk of arrhythmic events in pediatric patients with CPVT with and without an ICD. METHODS: We compared the risk of SCD in patients with RYR2 (ryanodine receptor 2) variants and phenotype-positive symptomatic CPVT patients with and without an ICD who were younger than 19 years and had no history of sudden cardiac arrest at phenotype diagnosis. The primary outcome was SCD; secondary outcomes were composite end points of SCD, sudden cardiac arrest, or appropriate ICD shocks with or without arrhythmic syncope. RESULTS: The study included 235 patients, 73 with an ICD (31.1%) and 162 without an ICD (68.9%). Over a median follow-up of 8.0 years (interquartile range 4.3-13.4 years), SCD occurred in 7 patients (3.0%), of whom 4 (57.1%) were noncompliant with medications and none had an ICD. Patients with ICD had a higher risk of both secondary composite outcomes (without syncope: hazard ratio 5.85; 95% confidence interval 3.40-10.09; P < .0001; with syncope: hazard ratio 2.55; 95% confidence interval 1.50-4.34; P = .0005). Thirty-one patients with ICD (42.5%) experienced appropriate shocks, 18 (24.7%) inappropriate shocks, and 21 (28.8%) device-related complications. CONCLUSION: SCD events occurred only in patients without an ICD and mostly in those not on optimal medical therapy. Patients with an ICD had a high risk of appropriate and inappropriate shocks, which may be reduced with appropriate device programming. Severe ICD complications were common, and risks vs benefits of ICDs need to be considered.
Cardiology Service Hospital Josep Trueta Girona Spain
Centro de Investigación Biomédica en Red de Enfermedades Cardiovasculares Madrid Spain
Children's Hospital Los Angeles Los Angeles California
Department of Cardiology Royal Brompton Hospital London UK
Department of Cardiovascular Diseases University Hospitals Leuven Belgium
Department of Cardiovascular Medicine National Cerebral and Cardiovascular Centre Suita Japan
Department of Cardiovascular Medicine Shiga University of Medical Science Otsu Japan
Department of Cardiovascular Sciences University of Leuven Leuven Belgium
Department of Medicine University Medical Center Mannheim Mannheim Germany
Department of Paediatrics Child and Youth Health The University of Auckland Auckland New Zealand
Department of Pediatric Cardiology Erasmus MC Sophia Rotterdam The Netherlands
Faculty of Medicine and Health The University of Sydney Sydney Australia
German Center for Cardiovascular Research Partner Site Heidelberg Mannheim Germany
Heart and Lung Centre Helsinki University Hospital and Helsinki University Helsinki Finland
Hong Kong Children's Hospital Hong Kong SAR China
Istituto Auxologico Italiano IRCCS Center for Cardiac Arrhythmias of Genetic Origin Milan Italy
Medical Science Department School of Medicine Universitat de Girona Girona Spain
Medical Science Department School of Medicine University of Girona Girona Spain
Nationwide Children's Hospital Columbus Ohio
Nemours Children's Clinic Orlando Florida
Rady Children's Hospital San Diego California
Sibley Heart Center Children's Healthcare of Atlanta Atlanta Georgia
Université de Nantes CHU Nantes CNRS INSERM l'institut du thorax Nantes France
University of Gottingen Gottingen Germany
University of Iowa Stead Family Children's Hospital Iowa City Iowa
References provided by Crossref.org
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- $a An international multicenter cohort study on implantable cardioverter-defibrillators for the treatment of symptomatic children with catecholaminergic polymorphic ventricular tachycardia / $c A. Lamba, TM. Roston, PJ. Peltenburg, D. Kallas, S. Franciosi, KVV. Lieve, PJ. Kannankeril, M. Horie, S. Ohno, R. Brugada, T. Aiba, P. Fischbach, L. Knight, J. Till, SY. Kwok, V. Probst, D. Backhoff, MJ. LaPage, AS. Batra, F. Drago, K. Haugaa, AD. Krahn, T. Robyns, H. Swan, T. Tavacova, C. van der Werf, J. Atallah, M. Borggrefe, B. Rudic, G. Sarquella-Brugada, E. Chorin, A. Hill, J. Kammeraad, A. Kamp, I. Law, J. Perry, JD. Roberts, S. Tisma-Dupanovic, C. Semsarian, JR. Skinner, J. Tfelt-Hansen, I. Denjoy, A. Leenhardt, PJ. Schwartz, MJ. Ackerman, NA. Blom, AAM. Wilde, S. Sanatani
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- $a BACKGROUND: Catecholaminergic polymorphic ventricular tachycardia (CPVT) may cause sudden cardiac death (SCD) despite medical therapy. Therefore, implantable cardioverter-defibrillators (ICDs) are commonly advised. However, there is limited data on the outcomes of ICD use in children. OBJECTIVE: The purpose of this study was to compare the risk of arrhythmic events in pediatric patients with CPVT with and without an ICD. METHODS: We compared the risk of SCD in patients with RYR2 (ryanodine receptor 2) variants and phenotype-positive symptomatic CPVT patients with and without an ICD who were younger than 19 years and had no history of sudden cardiac arrest at phenotype diagnosis. The primary outcome was SCD; secondary outcomes were composite end points of SCD, sudden cardiac arrest, or appropriate ICD shocks with or without arrhythmic syncope. RESULTS: The study included 235 patients, 73 with an ICD (31.1%) and 162 without an ICD (68.9%). Over a median follow-up of 8.0 years (interquartile range 4.3-13.4 years), SCD occurred in 7 patients (3.0%), of whom 4 (57.1%) were noncompliant with medications and none had an ICD. Patients with ICD had a higher risk of both secondary composite outcomes (without syncope: hazard ratio 5.85; 95% confidence interval 3.40-10.09; P < .0001; with syncope: hazard ratio 2.55; 95% confidence interval 1.50-4.34; P = .0005). Thirty-one patients with ICD (42.5%) experienced appropriate shocks, 18 (24.7%) inappropriate shocks, and 21 (28.8%) device-related complications. CONCLUSION: SCD events occurred only in patients without an ICD and mostly in those not on optimal medical therapy. Patients with an ICD had a high risk of appropriate and inappropriate shocks, which may be reduced with appropriate device programming. Severe ICD complications were common, and risks vs benefits of ICDs need to be considered.
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- $a Peltenburg, Puck J $u Amsterdam UMC, University of Amsterdam, Heart Centre, Department of Clinical and Experimental Cardiology, Amsterdam Cardiovascular Sciences, Heart Failure and Arrhythmias, Amsterdam, the Netherlands; Department of Pediatric Cardiology, Amsterdam UMC, University of Amsterdam, Emma Children's Hospital, Amsterdam, The Netherlands
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