Sudden cardiac arrest in infants and children: proposal for a diagnostic workup to identify the etiology. An 18-year multicenter evaluation in the Netherlands.
Cardiac history
Diagnostic workup
Etiologies
Pediatrics
Sudden cardiac arrest
Journal
European journal of pediatrics
ISSN: 1432-1076
Titre abrégé: Eur J Pediatr
Pays: Germany
ID NLM: 7603873
Informations de publication
Date de publication:
Jan 2024
Jan 2024
Historique:
received:
22
08
2023
accepted:
21
10
2023
revised:
17
10
2023
medline:
9
2
2024
pubmed:
27
10
2023
entrez:
27
10
2023
Statut:
ppublish
Résumé
Sudden cardiac arrest (SCA) studies are often population-based, limited to sudden cardiac death, and excluding infants. To guide prevention opportunities, it is essential to be informed of pediatric SCA etiologies. Unfortunately, etiologies frequently remain unresolved. The objectives of this study were to determine paediatric SCA etiology, and to evaluate the extent of post-SCA investigations and to assess the performance of previous cardiac evaluation in detecting conditions predisposing to SCA. In a retrospective cohort (2002-2019), all children 0-18 years with out-of-hospital cardiac arrest (OHCA) referred to Erasmus MC Sophia Children's Hospital or the Amsterdam UMC (tertiary-care university hospitals), with cardiac or unresolved etiologies were eligible for inclusion. SCA etiologies, cardiac and family history and etiologic investigations in unresolved cases were assessed. The etiology of arrest could be determined in 52% of 172 cases. Predominant etiologies in children ≥ 1 year (n = 99) were primary arrhythmogenic disorders (34%), cardiomyopathies (22%) and unresolved (32%). Events in children < 1 year (n = 73) were largely unresolved (70%) or caused by cardiomyopathy (8%), congenital heart anomaly (8%) or myocarditis (7%). Of 83 children with unresolved etiology a family history was performed in 51%, an autopsy in 51% and genetic testing in 15%. Pre-existing cardiac conditions presumably causative for SCA were diagnosed in 9%, and remained unrecognized despite prior evaluation in 13%. SCA etiology remained unresolved in 83 of 172 cases (48%) and essential diagnostic investigations were often not performed. Over one-fifth of SCA patients underwent prior cardiac evaluation, which did not lead to recognition of a cardiac condition predisposing to SCA in all of them. The diagnostic post-SCA approach should be improved and the proposed standardized pediatric post-SCA diagnostics protocol may ensure a consistent and systematic evaluation process increasing the diagnostic yield. • Arrests in infants remain unresolved in most cases. In children > 1 year, predominant etiologies are primary arrhythmia disorders, cardiomyopathy and myocarditis. • Studies investigating sudden cardiac arrest are often limited to sudden cardiac death (SCD) in 1 to 40 year old persons, excluding infants and successfully resuscitated children. • In patients with unresolved SCA events, the diagnostic work up was often incompletely performed. • Over one fifth of victims had prior cardiac evaluation before the arrest, with either a diagnosed cardiac condition (9%) or an unrecognized cardiac condition (13%).
Identifiants
pubmed: 37889292
doi: 10.1007/s00431-023-05301-9
pii: 10.1007/s00431-023-05301-9
pmc: PMC10858117
doi:
Types de publication
Multicenter Study
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
335-344Informations de copyright
© 2023. The Author(s).
Références
Bardai A, Berdowski J, van der Werf C, Blom MT, Ceelen M, van Langen IM et al (2011) Incidence, causes, and outcomes of out-of-hospital cardiac arrest in children. A comprehensive, prospective, population-based study in the Netherlands. J Am Coll Cardiol 57(18):1822–1828
pubmed: 21527156
doi: 10.1016/j.jacc.2010.11.054
Hunfeld M, Dulfer K, Rietman A, Pangalila R, van Gils-Frijters A, Catsman-Berrevoets C et al (2021) Longitudinal two years evaluation of neuropsychological outcome in children after out of hospital cardiac arrest. Resuscitation 167:29–37
pubmed: 34389455
doi: 10.1016/j.resuscitation.2021.07.043
Fink EL, Prince DK, Kaltman JR, Atkins DL, Austin M, Warden C et al (2016) Unchanged pediatric out-of-hospital cardiac arrest incidence and survival rates with regional variation in North America. Resuscitation 107:121–128
pubmed: 27565862
pmcid: 5037038
doi: 10.1016/j.resuscitation.2016.07.244
Bagnall RD, Weintraub RG, Ingles J, Duflou J, Yeates L, Lam L et al (2016) A prospective study of sudden cardiac death among children and young adults. N Engl J Med 374(25):2441–2452
pubmed: 27332903
doi: 10.1056/NEJMoa1510687
Maron BJ, Doerer JJ, Haas TS, Tierney DM, Mueller FO (2009) Sudden deaths in young competitive athletes: analysis of 1866 deaths in the United States, 1980–2006. Circulation 119(8):1085–1092
pubmed: 19221222
doi: 10.1161/CIRCULATIONAHA.108.804617
Winkel BG, Holst AG, Theilade J, Kristensen IB, Thomsen JL, Ottesen GL et al (2011) Nationwide study of sudden cardiac death in persons aged 1–35 years. Eur Heart J 32(8):983–990
pubmed: 21131293
doi: 10.1093/eurheartj/ehq428
Meyer L, Stubbs B, Fahrenbruch C, Maeda C, Harmon K, Eisenberg M et al (2012) Incidence, causes, and survival trends from cardiovascular-related sudden cardiac arrest in children and young adults 0 to 35 years of age: a 30-year review. Circulation 126(11):1363–1372
pubmed: 22887927
doi: 10.1161/CIRCULATIONAHA.111.076810
Virani SS, Alonso A, Benjamin EJ, Bittencourt MS, Callaway CW, Carson AP et al (2020) Heart disease and stroke statistics-2020 update: a report from the American Heart Association. Circulation 141(9):e139–e596
pubmed: 31992061
doi: 10.1161/CIR.0000000000000757
Lynge TH, Nielsen JL, Risgaard B, van der Werf C, Winkel BG, Tfelt-Hansen J (2023) Causes of sudden cardiac death according to age and sex in persons aged 1–49 years. Heart Rhythm 20(1):61–68
pubmed: 36075534
doi: 10.1016/j.hrthm.2022.08.036
Liebrechts-Akkerman G, Liu F, van Marion R, Dinjens WNM, Kayser M (2020) Explaining sudden infant death with cardiac arrhythmias: Complete exon sequencing of nine cardiac arrhythmia genes in Dutch SIDS cases highlights new and known DNA variants. Forensic Sci Int Genet 46:102266
pubmed: 32145446
doi: 10.1016/j.fsigen.2020.102266
Tester DJ, Wong LCH, Chanana P, Jaye A, Evans JM, FitzPatrick DR et al (2018) Cardiac genetic predisposition in sudden infant death syndrome. J Am Coll Cardiol 71(11):1217–1227
pubmed: 29544605
doi: 10.1016/j.jacc.2018.01.030
Glinge C, Rossetti S, Oestergaard LB, Stampe NK, Lynge TH, Skals R et al (2023) Risk of sudden infant death syndrome among siblings of children who died of sudden infant death syndrome in Denmark. JAMA Netw Open 6(1):e2252724
pubmed: 36696110
pmcid: 10187488
doi: 10.1001/jamanetworkopen.2022.52724
Baruteau AE, Tester DJ, Kapplinger JD, Ackerman MJ, Behr ER (2017) Sudden infant death syndrome and inherited cardiac conditions. Nat Rev Cardiol 14(12):715–726
pubmed: 28880023
doi: 10.1038/nrcardio.2017.129
Tester DJ, Wong LCH, Chanana P, Gray B, Jaye A, Evans JM et al (2018) Exome-wide rare variant analyses in sudden infant death syndrome. J Pediatr 203(423–8):e11
Adabag AS, Luepker RV, Roger VL, Gersh BJ (2010) Sudden cardiac death: epidemiology and risk factors. Nat Rev Cardiol 7(4):216–225
pubmed: 20142817
pmcid: 5014372
doi: 10.1038/nrcardio.2010.3
Semsarian C, Ingles J, Wilde AA (2015) Sudden cardiac death in the young: the molecular autopsy and a practical approach to surviving relatives. Eur Heart J 36(21):1290–1296
pubmed: 25765769
doi: 10.1093/eurheartj/ehv063
Wilde AAM, Semsarian C, Marquez MF, Sepehri Shamloo A, Ackerman MJ, Ashley EA et al (2022) European Heart Rhythm Association (EHRA)/Heart Rhythm Society (HRS)/Asia Pacific Heart Rhythm Society (APHRS)/Latin American Heart Rhythm Society (LAHRS) Expert Consensus Statement on the state of genetic testing for cardiac diseases. Heart Rhythm 19(7):e1–e60
pubmed: 35390533
doi: 10.1016/j.hrthm.2022.03.1225
Stiles MK, Wilde AAM, Abrams DJ, Ackerman MJ, Albert CM, Behr ER et al (2021) 2020 APHRS/HRS expert consensus statement on the investigation of decedents with sudden unexplained death and patients with sudden cardiac arrest, and of their families. Heart Rhythm 18(1):e1–e50
pubmed: 33091602
doi: 10.1016/j.hrthm.2020.10.010
Cirino AL, Ho CY (2013) Genetic testing for inherited heart disease. Circulation 128(1):e4–8
pubmed: 23817488
pmcid: 3769178
doi: 10.1161/CIRCULATIONAHA.113.002252
Tan HL, Hofman N, van Langen IM, van der Wal AC, Wilde AA (2005) Sudden unexplained death: heritability and diagnostic yield of cardiological and genetic examination in surviving relatives. Circulation 112(2):207–213
pubmed: 15998675
doi: 10.1161/CIRCULATIONAHA.104.522581
Campell R, Berger S, Ackerman MJ, Morrow WR, Jenkins K, Minich LL et al (2012) Pediatric sudden cardiac arrest. Pediatrics 129(4):e1094–e1102
doi: 10.1542/peds.2012-0144
Albrecht M, de Jonge RCJ, Nadkarni VM, de Hoog M, Hunfeld M, Kammeraad JAE et al (2021) Association between shockable rhythms and long-term outcome after pediatric out-of-hospital cardiac arrest in Rotterdam, the Netherlands: An 18-year observational study. Resuscitation 166:110–120
pubmed: 34082030
doi: 10.1016/j.resuscitation.2021.05.015
Priori SG, Wilde AA, Horie M, Cho Y, Behr ER, Berul C et al (2013) Executive summary: HRS/EHRA/APHRS expert consensus statement on the diagnosis and management of patients with inherited primary arrhythmia syndromes. Europace 15(10):1389–1406
pubmed: 23994779
doi: 10.1093/europace/eut272
Alapati S, Strobel N, Hashmi S, Bricker JT, Gupta-Malhotra M (2013) Sudden unexplained cardiac arrest in apparently healthy children: a single-center experience. Pediatr Cardiol 34(3):639–645
pubmed: 23052663
doi: 10.1007/s00246-012-0516-0
Chugh SS, Reinier K, Balaji S, Uy-Evanado A, Vickers C, Mariani R et al (2009) Population-based analysis of sudden death in children: The Oregon Sudden Unexpected Death Study. Heart Rhythm 6(11):1618–1622
pubmed: 19879540
pmcid: 2803115
doi: 10.1016/j.hrthm.2009.07.046
Sweeting J, Semsarian C (2014) Cardiac abnormalities and sudden infant death syndrome. Paediatr Respir Rev 15(4):301–306
pubmed: 25301030
Hofman N, Tan HL, Alders M, Kolder I, de Haij S, Mannens MM et al (2013) Yield of molecular and clinical testing for arrhythmia syndromes: report of 15 years’ experience. Circulation 128(14):1513–1521
pubmed: 23963746
doi: 10.1161/CIRCULATIONAHA.112.000091
Sharifzadehgan A, Gaye B, Bougouin W, Narayanan K, Dumas F, Karam N et al (2022) Lack of early etiologic investigations in young sudden cardiac death. Resuscitation 179:197–205
pubmed: 35788021
doi: 10.1016/j.resuscitation.2022.06.023
Jarman JW, Lascelles K, Wong T, Markides V, Clague JR, Till J (2012) Clinical experience of entirely subcutaneous implantable cardioverter-defibrillators in children and adults: cause for caution. Eur Heart J 33(11):1351–1359
pubmed: 22408031
doi: 10.1093/eurheartj/ehs017
Priori SG, Wilde AA, Horie M, Cho Y, Behr ER, Berul C et al (2013) HRS/EHRA/APHRS expert consensus statement on the diagnosis and management of patients with inherited primary arrhythmia syndromes: document endorsed by HRS, EHRA, and APHRS in May 2013 and by ACCF, AHA, PACES, and AEPC in June 2013. Heart Rhythm 10(12):1932–1963
pubmed: 24011539
doi: 10.1016/j.hrthm.2013.05.014
Priori SG, Blomstrom-Lundqvist C, Mazzanti A, Blom N, Borggrefe M, Camm J et al (2015) Guidelines for the management of patients with ventricular arrhythmias and the prevention of sudden cardiac death: The Task Force for the Management of Patients with Ventricular Arrhythmias and the Prevention of Sudden Cardiac Death of the European Society of Cardiology (ESC). Endorsed by: Association for European Paediatric and Congenital Cardiology (AEPC). Eur Heart J 36(41):2793–2867
pubmed: 26320108
doi: 10.1093/eurheartj/ehv316
Pilmer CM, Kirsh JA, Hildebrandt D, Krahn AD, Gow RM (2014) Sudden cardiac death in children and adolescents between 1 and 19 years of age. Heart Rhythm 11(2):239–245
pubmed: 24239636
doi: 10.1016/j.hrthm.2013.11.006
Winkel BG, Risgaard B, Sadjadieh G, Bundgaard H, Haunso S, Tfelt-Hansen J (2014) Sudden cardiac death in children (1–18 years): symptoms and causes of death in a nationwide setting. Eur Heart J 35(13):868–875
pubmed: 24344190
doi: 10.1093/eurheartj/eht509
Bardai A, Blom MT, van Noord C, Verhamme KM, Sturkenboom MC, Tan HL (2015) Sudden cardiac death is associated both with epilepsy and with use of antiepileptic medications. Heart 101(1):17–22
pubmed: 25031263
doi: 10.1136/heartjnl-2014-305664
Chahal CAA, Salloum MN, Alahdab F, Gottwald JA, Tester DJ, Anwer LA et al (2020) Systematic review of the genetics of sudden unexpected death in epilepsy: potential overlap with sudden cardiac death and arrhythmia-related genes. J Am Heart Assoc 9(1):e012264
pubmed: 31865891
doi: 10.1161/JAHA.119.012264
Johnson JN, Hofman N, Haglund CM, Cascino GD, Wilde AA, Ackerman MJ (2009) Identification of a possible pathogenic link between congenital long QT syndrome and epilepsy. Neurology 72(3):224–231
pubmed: 19038855
pmcid: 2677528
doi: 10.1212/01.wnl.0000335760.02995.ca
MacCormick JM, McAlister H, Crawford J, French JK, Crozier I, Shelling AN et al (2009) Misdiagnosis of long QT syndrome as epilepsy at first presentation. Ann Emerg Med 54(1):26–32
pubmed: 19282063
doi: 10.1016/j.annemergmed.2009.01.031
Goldenberg I, Horr S, Moss AJ, Lopes CM, Barsheshet A, McNitt S et al (2011) Risk for life-threatening cardiac events in patients with genotype-confirmed long-QT syndrome and normal-range corrected QT intervals. J Am Coll Cardiol 57(1):51–59
pubmed: 21185501
pmcid: 3332533
doi: 10.1016/j.jacc.2010.07.038
Roston TM, Yuchi Z, Kannankeril PJ, Hathaway J, Vinocur JM, Etheridge SP et al (2018) The clinical and genetic spectrum of catecholaminergic polymorphic ventricular tachycardia: findings from an international multicentre registry. Europace 20(3):541–547
pubmed: 28158428
doi: 10.1093/europace/euw389
Peltenburg PJ, Hoedemaekers YM, Clur SAB, Blom NA, Blank AC, Boesaard EP et al (2023) Screening, diagnosis and follow-up of Brugada syndrome in children: a Dutch expert consensus statement. Neth Heart J 31(4):133–137
pubmed: 36223066
doi: 10.1007/s12471-022-01723-6
Naim MY, Burke RV, McNally BF, Song L, Griffis HM, Berg RA et al (2017) Association of bystander cardiopulmonary resuscitation with overall and neurologically favorable survival after pediatric out-of-hospital cardiac arrest in the United States: a report from the cardiac arrest registry to enhance survival surveillance registry. JAMA Pediatr 171(2):133–141
pubmed: 27837587
doi: 10.1001/jamapediatrics.2016.3643