{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,28]],"date-time":"2026-10-28T20:04:05Z","timestamp":1793217845478,"version":"4.5.0"},"reference-count":23,"publisher":"Oxford University Press (OUP)","issue":"35","license":[{"start":{"date-parts":[[2019,5,30]],"date-time":"2019-05-30T00:00:00Z","timestamp":1559174400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"funder":[{"name":"ZonMW Priority Medicines for Rare Diseases and Orphan Drugs","award":["113304045"],"award-info":[{"award-number":["113304045"]}]},{"DOI":"10.13039\/501100000925","name":"National Health and Medical Research Council","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100000925","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100000925","name":"NHMRC","doi-asserted-by":"publisher","award":["1059156"],"award-info":[{"award-number":["1059156"]}],"id":[{"id":"10.13039\/501100000925","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Mayo Clinic Windland Smith Rice Comprehensive Sudden Cardiac Death Program"},{"DOI":"10.13039\/501100012524","name":"Netherlands Federation of University Medical Centres","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100012524","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001826","name":"Netherlands Organisation for Health Research and Development","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100001826","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001722","name":"Royal Netherlands Academy of Sciences","doi-asserted-by":"crossref","award":["CVON 2012-10 PREDICT"],"award-info":[{"award-number":["CVON 2012-10 PREDICT"]}],"id":[{"id":"10.13039\/501100001722","id-type":"DOI","asserted-by":"crossref"}]},{"name":"E-Rare Joint Transnational Call for Proposals 2015 \u2018Improving Diagnosis and Treatment of Catecholaminergic Polymorphic Ventricular Tachycardia: Integrating Clinical and Basic Science\u2019"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2019,9,14]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Aims<\/jats:title>\n                    <jats:p>In patients with catecholaminergic polymorphic ventricular tachycardia (CPVT), implantable cardioverter-defibrillator (ICD) shocks are sometimes ineffective and may even trigger fatal electrical storms. We assessed the efficacy and complications of ICDs placed in patients with CPVT who presented with a sentinel event of sudden cardiac arrest (SCA) while undiagnosed and therefore untreated.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods and results<\/jats:title>\n                    <jats:p>We analysed 136 patients who presented with SCA and in whom CPVT was diagnosed subsequently, leading to the initiation of guideline-directed therapy, including \u03b2-blockers, flecainide, and\/or left cardiac sympathetic denervation. An ICD was implanted in 79 patients (58.1%). The primary outcome of the study was sudden cardiac death (SCD). The secondary outcomes were composite outcomes of SCD, SCA, appropriate ICD shocks, and syncope. After a median follow-up of 4.8\u2009years, SCD had occurred in three patients (3.8%) with an ICD and none of the patients without an ICD (P\u2009=\u20090.1). SCD, SCA, or appropriate ICD shocks occurred in 37 patients (46.8%) with an ICD and 9 patients (15.8%) without an ICD (P\u2009&amp;lt;\u20090.0001). Inappropriate ICD shocks occurred in 19 patients (24.7%) and other device-related complications in 22 patients (28.9%).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>In previously undiagnosed patients with CPVT who presented with SCA, an ICD was not associated with improved survival. Instead, the ICD was associated with both a high rate of appropriate ICD shocks and inappropriate ICD shocks along with other device-related complications. Strict adherence to guideline-directed therapy without an ICD may provide adequate protection in these patients without all the potential disadvantages of an ICD.<\/jats:p>\n                    <jats:p\/>\n                  <\/jats:sec>","DOI":"10.1093\/eurheartj\/ehz309","type":"journal-article","created":{"date-parts":[[2019,4,26]],"date-time":"2019-04-26T23:35:41Z","timestamp":1556321741000},"page":"2953-2961","source":"Crossref","is-referenced-by-count":133,"title":["Implantable cardioverter-defibrillators in previously undiagnosed patients with catecholaminergic polymorphic ventricular tachycardia resuscitated from sudden cardiac arrest"],"prefix":"10.1093","volume":"40","author":[{"given":"Christian","family":"van der Werf","sequence":"first","affiliation":[{"name":"Amsterdam UMC, University of Amsterdam, Heart Centre, and Department of Clinical and Experimental Cardiology, Amsterdam Cardiovascular Sciences, Meibergdreef 9 , 1105 AZ Amsterdam, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Krystien V","family":"Lieve","sequence":"additional","affiliation":[{"name":"Amsterdam UMC, University of Amsterdam, Heart Centre, and Department of Clinical and Experimental Cardiology, Amsterdam Cardiovascular Sciences, Meibergdreef 9 , 1105 AZ Amsterdam, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J Martijn","family":"Bos","sequence":"additional","affiliation":[{"name":"Division of Heart Rhythm Services, Department of Cardiovascular Medicine, Mayo Clinic, 200 First St SW , Rochester, MN, USA"},{"name":"Division of Pediatric Cardiology, Department of Pediatric and Adolescent Medicine, Mayo Clinic, 200 First St SW , Rochester, MN, USA"},{"name":"Department of Molecular Pharmacology and Experimental Therapeutics, Windland Smith Rice Sudden Death Genomics Laboratory, Mayo Clinic, 200 First St SW , Rochester, MN, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Conor M","family":"Lane","sequence":"additional","affiliation":[{"name":"Division of Heart Rhythm Services, Department of Cardiovascular Medicine, Mayo Clinic, 200 First St SW , Rochester, MN, USA"},{"name":"Division of Pediatric Cardiology, Department of Pediatric and Adolescent Medicine, Mayo Clinic, 200 First St SW , Rochester, MN, USA"},{"name":"Department of Molecular Pharmacology and Experimental Therapeutics, Windland Smith Rice Sudden Death Genomics Laboratory, Mayo Clinic, 200 First St SW , Rochester, MN, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Isabelle","family":"Denjoy","sequence":"additional","affiliation":[{"name":"Service de Cardiologie et CNMR Maladies Cardiaques H\u00e9r\u00e9ditaires Rares, H\u00f4pital Bichat, 46 Rue Henri Huchard , Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5955-7490","authenticated-orcid":false,"given":"Ferran","family":"Roses-Noguer","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Royal Brompton Hospital, Sydney S t, Chelsea, London, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Takeshi","family":"Aiba","sequence":"additional","affiliation":[{"name":"Division of Arrhythmia and Electrophysiology, Department of Cardiovascular Medicine, National Cerebral and Cardiovascular Centre, 5 Chome-7-1 Fujishirodai , Suita, Osaka, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8533-8040","authenticated-orcid":false,"given":"Yuko","family":"Wada","sequence":"additional","affiliation":[{"name":"Department of Cardiovascular Medicine, Shiga University of Medical Science, Seta Tsukinowacho , Otsu, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4846-7676","authenticated-orcid":false,"given":"Jodie","family":"Ingles","sequence":"additional","affiliation":[{"name":"Agnes Ginges Centre for Molecular Cardiology at Centenary Institute, The University of Sydney, Locked Bag 6 , Newtown, Sydney, Australia"},{"name":"Faculty of Medicine and Health, The University of Sydney, Locked Bag 6, Newtown , Sydney, Australia"},{"name":"Department of Cardiology, Royal Prince Alfred Hospital, Locked Bag 6, Newtown , Sydney, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2716-9178","authenticated-orcid":false,"given":"Ida S","family":"Leren","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Centre for Cardiological Innovation, Oslo University Hospital, Rikshospitalet , Sognsvannsveien 20, Oslo, Norway"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Boris","family":"Rudic","sequence":"additional","affiliation":[{"name":"Department of Cardiology, University Medical Centre Mannheim, Theodor-Kutzer-Ufer 1 - 3 , Mannheim, Germany"},{"name":"German Centre for Cardiovascular Research (DZHK), Partner Site Heidelberg\/Mannheim , Mannheim, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0367-1048","authenticated-orcid":false,"given":"Peter J","family":"Schwartz","sequence":"additional","affiliation":[{"name":"Istituto Auxologico Italiano, IRCCS, Center for Cardiac Arrhythmias of Genetic Origin, Via Pier Lombardo 22 , Milan, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9934-6746","authenticated-orcid":false,"given":"Alice","family":"Maltret","sequence":"additional","affiliation":[{"name":"H\u00f4pital Necker-Enfants-Malades, Cardiologie P\u00e9diatrique, 149 Rue de S\u00e8vres , Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frederic","family":"Sacher","sequence":"additional","affiliation":[{"name":"LIRYC Institute, Bordeaux University Hospital, Bordeaux University, Avenue du Haut L\u00e9v\u00eaque , Pessac- Bordeaux, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3653-0412","authenticated-orcid":false,"given":"Jonathan R","family":"Skinner","sequence":"additional","affiliation":[{"name":"Cardiac Inherited Disease Group New Zealand, Green Lane Paediatric and Congenital Cardiac Services, Starship Children's Hospital , 2 Park Rd, Grafton, Auckland 1023 New Zealand"},{"name":"Department of Paediatrics Child and Youth Health, The University of Auckland , Auckland, New Zealand"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7982-1586","authenticated-orcid":false,"given":"Andrew D","family":"Krahn","sequence":"additional","affiliation":[{"name":"Division of Cardiology, Heart Rhythm Services, University of British Columbia , 1033 Davie Street, Vancouver, BC, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5663-4377","authenticated-orcid":false,"given":"Thomas M","family":"Roston","sequence":"additional","affiliation":[{"name":"Division of Cardiology, Heart Rhythm Services, University of British Columbia , 1033 Davie Street, Vancouver, BC, Canada"},{"name":"BC Children's Hospital, 4480 Oak St , Vancouver, BC, Canada"},{"name":"Department of Pediatrics, University of British Columbia , 4480 Oak Street, Vancouver, BC, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jacob","family":"Tfelt-Hansen","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Rigshospitalet, Blegdamsvej 9 , Copenhagen, Denmark"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Heikki","family":"Swan","sequence":"additional","affiliation":[{"name":"Heart and Lung Centre, Helsinki University Hospital and Helsinki University , Tukholmankatu 8 A Helsinki, Finland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8676-4874","authenticated-orcid":false,"given":"Tomas","family":"Robyns","sequence":"additional","affiliation":[{"name":"Department of Cardiovascular Diseases, University Hospitals Leuven , Herestraat 49, Leuven, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Seiko","family":"Ohno","sequence":"additional","affiliation":[{"name":"Department of Cardiovascular Medicine, Shiga University of Medical Science, Seta Tsukinowacho , Otsu, Japan"},{"name":"Department of Bioscience and Genetics, National Cerebral and Cardiovascular Centre , 5 Chome-7-1 Fujishirodai, Suita, Osaka, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jason D","family":"Roberts","sequence":"additional","affiliation":[{"name":"Section of Cardiac Electrophysiology, Division of Cardiology, Department of Medicine, Western University , 339 Windermere Road, B6-129B, London, ON, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maarten P","family":"van den Berg","sequence":"additional","affiliation":[{"name":"Department of Cardiology, University of Groningen, University Medical Centre Groningen, Hanzeplein 1 , GZ Groningen, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Janneke A","family":"Kammeraad","sequence":"additional","affiliation":[{"name":"Department of Pediatric Cardiology, Sophia Children\u2019s Hospital, Erasmus University Medical Centre, Doctor Molewaterplein 40 , 3015 GD Rotterdam, The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vincent","family":"Probst","sequence":"additional","affiliation":[{"name":"L'Institut du Thorax, Cardiologic Department and Reference Center for Hereditary Arrhythmic Diseases INSERM 1087 , Boulevard Monod, Nantes, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1529-9872","authenticated-orcid":false,"given":"Prince J","family":"Kannankeril","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Monroe Carell Jr Children's Hospital at Vanderbilt, Vanderbilt University Medical Centre, 2200 Children's Way , Nashville, TN, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nico A","family":"Blom","sequence":"additional","affiliation":[{"name":"Department of Pediatric Cardiology, Emma Children's Hospital, Amsterdam UMC, University of Amsterdam, Meibergdreef 9 , AZ Amsterdam, The Netherlands"},{"name":"Department of Pediatric Cardiology, Willem-Alexander Children's Hospital, Leiden University Medical Centre, Albinusdreef 2, ZA Leiden , The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8731-2853","authenticated-orcid":false,"given":"Elijah R","family":"Behr","sequence":"additional","affiliation":[{"name":"Molecular and Clinical Sciences Research Institute, St. George's, University of London, Cranmer Terrace , London, UK"},{"name":"Cardiology Clinical Academic Group, St. George\u2019s University Hospitals NHS Foundation Trust , Cranmer Terrace, London, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Martin","family":"Borggrefe","sequence":"additional","affiliation":[{"name":"Department of Cardiology, University Medical Centre Mannheim, Theodor-Kutzer-Ufer 1 - 3 , Mannheim, Germany"},{"name":"German Centre for Cardiovascular Research (DZHK), Partner Site Heidelberg\/Mannheim , Mannheim, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kristina H","family":"Haugaa","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Centre for Cardiological Innovation, Oslo University Hospital, Rikshospitalet , Sognsvannsveien 20, Oslo, Norway"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christopher","family":"Semsarian","sequence":"additional","affiliation":[{"name":"Agnes Ginges Centre for Molecular Cardiology at Centenary Institute, The University of Sydney, Locked Bag 6 , Newtown, Sydney, Australia"},{"name":"Faculty of Medicine and Health, The University of Sydney, Locked Bag 6, Newtown , Sydney, Australia"},{"name":"Department of Cardiology, Royal Prince Alfred Hospital, Locked Bag 6, Newtown , Sydney, Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9029-2339","authenticated-orcid":false,"given":"Minoru","family":"Horie","sequence":"additional","affiliation":[{"name":"Department of Cardiovascular Medicine, Shiga University of Medical Science, Seta Tsukinowacho , Otsu, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wataru","family":"Shimizu","sequence":"additional","affiliation":[{"name":"Division of Arrhythmia and Electrophysiology, Department of Cardiovascular Medicine, National Cerebral and Cardiovascular Centre, 5 Chome-7-1 Fujishirodai , Suita, Osaka, Japan"},{"name":"Department of Cardiovascular Medicine, Nippon Medical School, 1-1-5, Sendagi, Bunkyo-ku , Tokyo, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Janice A","family":"Till","sequence":"additional","affiliation":[{"name":"Department of Cardiology, Royal Brompton Hospital, Sydney S t, Chelsea, London, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Antoine","family":"Leenhardt","sequence":"additional","affiliation":[{"name":"Service de Cardiologie et CNMR Maladies Cardiaques H\u00e9r\u00e9ditaires Rares, H\u00f4pital Bichat, 46 Rue Henri Huchard , Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael J","family":"Ackerman","sequence":"additional","affiliation":[{"name":"Division of Heart Rhythm Services, Department of Cardiovascular Medicine, Mayo Clinic, 200 First St SW , Rochester, MN, USA"},{"name":"Division of Pediatric Cardiology, Department of Pediatric and Adolescent Medicine, Mayo Clinic, 200 First St SW , Rochester, MN, USA"},{"name":"Department of Molecular Pharmacology and Experimental Therapeutics, Windland Smith Rice Sudden Death Genomics Laboratory, Mayo Clinic, 200 First St SW , Rochester, MN, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0528-0852","authenticated-orcid":false,"given":"Arthur A","family":"Wilde","sequence":"additional","affiliation":[{"name":"Amsterdam UMC, University of Amsterdam, Heart Centre, and Department of Clinical and Experimental Cardiology, Amsterdam Cardiovascular Sciences, Meibergdreef 9 , 1105 AZ Amsterdam, The Netherlands"},{"name":"Brahim Centre of Excellence in Research of Hereditary Disorders, Princess Al-Jawhara Al, 7393 Al-Malae'b St, King Abdul Aziz University , Jeddah, Kingdom of Saudi Arabia"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2019,5,30]]},"reference":[{"key":"2022102609280249200_ehz309-B1","first-page":"1677","article-title":"2017 AHA\/ACC\/HRS Guideline for management of patients with ventricular arrhythmias and the prevention of sudden cardiac death: a report of the American College of Cardiology\/American Heart 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