Group A Streptococcal Disease in Sudden Unexpected Death in Youth in the Pre- and Post-COVID-19 Era

Evelien B. van Kempen; Annelotte M. Pries; Emmeline P. Buddingh; Patrycja J. Puiman; Mirjam van Veen; A. Custers; E. Edelenbos; J. Fuijkschot; B. Levelink; P. J. Puiman; J. M. Ruskamp; B. Semmekrot; K. T. Verbruggen; H. Vlaardingerbroek; Mirjam van Veen; Emmeline Buddingh; Evelien van Kempen; Adam Tulling; Erik von Asmuth; Ankie Lebon; Merijn Bijlsma; Nina van Sorge; Jan van der Linden; Jantien Bolt; Lonneke van Onzenoort-Bokken; Else Bijker; Dorine Borensztajn; Kim Stol; Brechje de Gier; Navin Boeddha; Rianne Oostenbrink; Marlies van Houten; Gerdien Tramper; Monique Jacobs; Caroline Kosterink-Brackel; Joanne Wildenbeest; PESUDY collaborative; COPP-IGAS collaborative

doi:10.1097/INF.0000000000004775

Group A Streptococcal Disease in Sudden Unexpected Death in Youth in the Pre- and Post-COVID-19 Era

Evelien B. van Kempen^*
, Annelotte M. Pries
, Emmeline P. Buddingh
, Patrycja J. Puiman
, Mirjam van Veen
, A. Custers
, E. Edelenbos
, J. Fuijkschot
, B. Levelink
, P. J. Puiman
, J. M. Ruskamp
, B. Semmekrot
, K. T. Verbruggen
, H. Vlaardingerbroek
, Mirjam van Veen
, Emmeline Buddingh
, Evelien van Kempen
, Adam Tulling
, Erik von Asmuth
, Ankie Lebon

Merijn Bijlsma, Nina van Sorge, Jan van der Linden, Jantien Bolt, Lonneke van Onzenoort-Bokken, Else Bijker, Dorine Borensztajn, Kim Stol, Brechje de Gier, Navin Boeddha, Rianne Oostenbrink, Marlies van Houten, Gerdien Tramper, Monique Jacobs, Caroline Kosterink-Brackel, Joanne Wildenbeest, PESUDY collaborative, COPP-IGAS collaborative

^*Corresponding author for this work

Erasmus University Rotterdam
Juliana Children's Hospital
Leiden University
Maastricht University
Amsterdam UMC - University of Amsterdam
Radboud University Nijmegen
Utrecht University
Canisius Wilhelmina Hospital
University of Groningen
Albert Schweitzer Ziekenhuis
Bern-hoven Hospital
Haaglanden Medisch Centrum
Maxima Medical Centre
Northwest Hospital Group
National Institute for Publich Health and the Environment
Maasstad Hospital
Spaarne Gasthuis
Sint-Franscisus Gasthuis Hospital
Slingeland Ziekenhuis
Tergooiziekenhuizen
Ziekenhuis De Heel, Zaandam

Research output: Contribution to journal › Article › Academic › peer-review

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Abstract

Background: An upsurge in pediatric invasive group A streptococcal infection (iGAS) has been observed in the Netherlands along with a suspected increase in iGAS-related sudden death. Sudden unexplained deaths in youth (SUDY) are investigated nationally through a standardized procedure [(Postmortem Evaluation of Sudden Unexplained Death in Youth (PESUDY)]. We investigate epidemiological differences between pediatric iGAS-related sudden deaths (iGAS-PESUDY) and surviving iGAS cases. Methods: This observational study used data from the COPP-iGAS study on pediatric iGAS infections in Dutch hospitals and the PESUDY database. Children 0–18 years of age were included between August 2016 and December 2022. Results: Twenty-one iGAS-PESUDY cases and 156 iGAS survivors were included. iGAS-PESUDY cases tended to be older compared to survivors. iGAS-PESUDY cases significantly increased in 2022 compared to the pre-COVID period. Pre- and/or coinciding infections were present in 66% of iGAS-PESUDY cases, predominantly varicella zoster (19%) and influenza (24%). In survivors, 13% had varicella zoster virus and 3% had influenza virus (P ≤ 0.001). C-reactive protein levels tended to be lower in iGAS-PESUDY cases (81 mg/L; interquartile range, 26.8–307.5) compared to survivors (266 mg/L; interquartile range, 218.0–302.0). Conclusion: iGAS is currently a prevalent cause of SUDY. The finding of moderately elevated C-reactive protein levels compared to high levels in survivors might suggest children dying suddenly of iGAS have a rapid and fulminant disease course. Children with a pre- and/or coinciding infection of varicella zoster or influenza virus may be at greater risk of succumbing to iGAS infections.

Original language	English
Pages (from-to)	e156-e160
Journal	Pediatric infectious disease journal
Volume	44
Issue number	5
DOIs	https://doi.org/10.1097/INF.0000000000004775
Publication status	Published - 1 May 2025

UN SDGs

This output contributes to the following UN Sustainable Development Goals (SDGs)

SDG 3 Good Health and Well-being

Keywords

epidemiology
group A streptococcus
sudden death

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van Kempen, E. B., Pries, A. M., Buddingh, E. P., Puiman, P. J., van Veen, M., Custers, A., Edelenbos, E., Fuijkschot, J., Levelink, B., Puiman, P. J., Ruskamp, J. M., Semmekrot, B., Verbruggen, K. T., Vlaardingerbroek, H., van Veen, M., Buddingh, E., van Kempen, E., Tulling, A., von Asmuth, E., ... COPP-IGAS collaborative (2025). Group A Streptococcal Disease in Sudden Unexpected Death in Youth in the Pre- and Post-COVID-19 Era. Pediatric infectious disease journal, 44(5), e156-e160. https://doi.org/10.1097/INF.0000000000004775

@article{7df11729045e4b32a4a3b1f08f2b8a78,

title = "Group A Streptococcal Disease in Sudden Unexpected Death in Youth in the Pre- and Post-COVID-19 Era",

abstract = "Background: An upsurge in pediatric invasive group A streptococcal infection (iGAS) has been observed in the Netherlands along with a suspected increase in iGAS-related sudden death. Sudden unexplained deaths in youth (SUDY) are investigated nationally through a standardized procedure [(Postmortem Evaluation of Sudden Unexplained Death in Youth (PESUDY)]. We investigate epidemiological differences between pediatric iGAS-related sudden deaths (iGAS-PESUDY) and surviving iGAS cases. Methods: This observational study used data from the COPP-iGAS study on pediatric iGAS infections in Dutch hospitals and the PESUDY database. Children 0–18 years of age were included between August 2016 and December 2022. Results: Twenty-one iGAS-PESUDY cases and 156 iGAS survivors were included. iGAS-PESUDY cases tended to be older compared to survivors. iGAS-PESUDY cases significantly increased in 2022 compared to the pre-COVID period. Pre- and/or coinciding infections were present in 66\% of iGAS-PESUDY cases, predominantly varicella zoster (19\%) and influenza (24\%). In survivors, 13\% had varicella zoster virus and 3\% had influenza virus (P ≤ 0.001). C-reactive protein levels tended to be lower in iGAS-PESUDY cases (81 mg/L; interquartile range, 26.8–307.5) compared to survivors (266 mg/L; interquartile range, 218.0–302.0). Conclusion: iGAS is currently a prevalent cause of SUDY. The finding of moderately elevated C-reactive protein levels compared to high levels in survivors might suggest children dying suddenly of iGAS have a rapid and fulminant disease course. Children with a pre- and/or coinciding infection of varicella zoster or influenza virus may be at greater risk of succumbing to iGAS infections.",

keywords = "epidemiology, group A streptococcus, sudden death",

author = "\{van Kempen\}, \{Evelien B.\} and Pries, \{Annelotte M.\} and Buddingh, \{Emmeline P.\} and Puiman, \{Patrycja J.\} and \{van Veen\}, Mirjam and A. Custers and E. Edelenbos and J. Fuijkschot and B. Levelink and Puiman, \{P. J.\} and Ruskamp, \{J. M.\} and B. Semmekrot and Verbruggen, \{K. T.\} and H. Vlaardingerbroek and \{van Veen\}, Mirjam and Emmeline Buddingh and \{van Kempen\}, Evelien and Adam Tulling and \{von Asmuth\}, Erik and Ankie Lebon and Merijn Bijlsma and \{van Sorge\}, Nina and \{van der Linden\}, Jan and Jantien Bolt and \{van Onzenoort-Bokken\}, Lonneke and Else Bijker and Dorine Borensztajn and Kim Stol and \{de Gier\}, Brechje and Navin Boeddha and Rianne Oostenbrink and \{van Houten\}, Marlies and Gerdien Tramper and Monique Jacobs and Caroline Kosterink-Brackel and Joanne Wildenbeest and \{PESUDY collaborative\} and Aline Verhage and \{COPP-IGAS collaborative\} and \{van der Aa\}, Leontien",

note = "Publisher Copyright: {\textcopyright} 2025 The Author(s).",

year = "2025",

month = may,

day = "1",

doi = "10.1097/INF.0000000000004775",

language = "English",

volume = "44",

pages = "e156--e160",

journal = "Pediatric infectious disease journal",

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van Kempen, EB, Pries, AM, Buddingh, EP, Puiman, PJ, van Veen, M, Custers, A, Edelenbos, E, Fuijkschot, J, Levelink, B, Puiman, PJ, Ruskamp, JM, Semmekrot, B, Verbruggen, KT, Vlaardingerbroek, H, van Veen, M, Buddingh, E, van Kempen, E, Tulling, A, von Asmuth, E, Lebon, A, Bijlsma, M , van Sorge, N, van der Linden, J, Bolt, J, van Onzenoort-Bokken, L, Bijker, E, Borensztajn, D, Stol, K, de Gier, B, Boeddha, N, Oostenbrink, R, van Houten, M, Tramper, G, Jacobs, M, Kosterink-Brackel, C, Wildenbeest, J, PESUDY collaborative & COPP-IGAS collaborative 2025, 'Group A Streptococcal Disease in Sudden Unexpected Death in Youth in the Pre- and Post-COVID-19 Era', Pediatric infectious disease journal, vol. 44, no. 5, pp. e156-e160. https://doi.org/10.1097/INF.0000000000004775

TY - JOUR

T1 - Group A Streptococcal Disease in Sudden Unexpected Death in Youth in the Pre- and Post-COVID-19 Era

AU - van Kempen, Evelien B.

AU - Pries, Annelotte M.

AU - Buddingh, Emmeline P.

AU - Puiman, Patrycja J.

AU - van Veen, Mirjam

AU - Custers, A.

AU - Edelenbos, E.

AU - Fuijkschot, J.

AU - Levelink, B.

AU - Puiman, P. J.

AU - Ruskamp, J. M.

AU - Semmekrot, B.

AU - Verbruggen, K. T.

AU - Vlaardingerbroek, H.

AU - van Veen, Mirjam

AU - Buddingh, Emmeline

AU - van Kempen, Evelien

AU - Tulling, Adam

AU - von Asmuth, Erik

AU - Lebon, Ankie

AU - Bijlsma, Merijn

AU - van Sorge, Nina

AU - van der Linden, Jan

AU - Bolt, Jantien

AU - van Onzenoort-Bokken, Lonneke

AU - Bijker, Else

AU - Borensztajn, Dorine

AU - Stol, Kim

AU - de Gier, Brechje

AU - Boeddha, Navin

AU - Oostenbrink, Rianne

AU - van Houten, Marlies

AU - Tramper, Gerdien

AU - Jacobs, Monique

AU - Kosterink-Brackel, Caroline

AU - Wildenbeest, Joanne

AU - PESUDY collaborative

AU - Verhage, Aline

AU - COPP-IGAS collaborative

AU - van der Aa, Leontien

PY - 2025/5/1

Y1 - 2025/5/1

N2 - Background: An upsurge in pediatric invasive group A streptococcal infection (iGAS) has been observed in the Netherlands along with a suspected increase in iGAS-related sudden death. Sudden unexplained deaths in youth (SUDY) are investigated nationally through a standardized procedure [(Postmortem Evaluation of Sudden Unexplained Death in Youth (PESUDY)]. We investigate epidemiological differences between pediatric iGAS-related sudden deaths (iGAS-PESUDY) and surviving iGAS cases. Methods: This observational study used data from the COPP-iGAS study on pediatric iGAS infections in Dutch hospitals and the PESUDY database. Children 0–18 years of age were included between August 2016 and December 2022. Results: Twenty-one iGAS-PESUDY cases and 156 iGAS survivors were included. iGAS-PESUDY cases tended to be older compared to survivors. iGAS-PESUDY cases significantly increased in 2022 compared to the pre-COVID period. Pre- and/or coinciding infections were present in 66% of iGAS-PESUDY cases, predominantly varicella zoster (19%) and influenza (24%). In survivors, 13% had varicella zoster virus and 3% had influenza virus (P ≤ 0.001). C-reactive protein levels tended to be lower in iGAS-PESUDY cases (81 mg/L; interquartile range, 26.8–307.5) compared to survivors (266 mg/L; interquartile range, 218.0–302.0). Conclusion: iGAS is currently a prevalent cause of SUDY. The finding of moderately elevated C-reactive protein levels compared to high levels in survivors might suggest children dying suddenly of iGAS have a rapid and fulminant disease course. Children with a pre- and/or coinciding infection of varicella zoster or influenza virus may be at greater risk of succumbing to iGAS infections.

AB - Background: An upsurge in pediatric invasive group A streptococcal infection (iGAS) has been observed in the Netherlands along with a suspected increase in iGAS-related sudden death. Sudden unexplained deaths in youth (SUDY) are investigated nationally through a standardized procedure [(Postmortem Evaluation of Sudden Unexplained Death in Youth (PESUDY)]. We investigate epidemiological differences between pediatric iGAS-related sudden deaths (iGAS-PESUDY) and surviving iGAS cases. Methods: This observational study used data from the COPP-iGAS study on pediatric iGAS infections in Dutch hospitals and the PESUDY database. Children 0–18 years of age were included between August 2016 and December 2022. Results: Twenty-one iGAS-PESUDY cases and 156 iGAS survivors were included. iGAS-PESUDY cases tended to be older compared to survivors. iGAS-PESUDY cases significantly increased in 2022 compared to the pre-COVID period. Pre- and/or coinciding infections were present in 66% of iGAS-PESUDY cases, predominantly varicella zoster (19%) and influenza (24%). In survivors, 13% had varicella zoster virus and 3% had influenza virus (P ≤ 0.001). C-reactive protein levels tended to be lower in iGAS-PESUDY cases (81 mg/L; interquartile range, 26.8–307.5) compared to survivors (266 mg/L; interquartile range, 218.0–302.0). Conclusion: iGAS is currently a prevalent cause of SUDY. The finding of moderately elevated C-reactive protein levels compared to high levels in survivors might suggest children dying suddenly of iGAS have a rapid and fulminant disease course. Children with a pre- and/or coinciding infection of varicella zoster or influenza virus may be at greater risk of succumbing to iGAS infections.

KW - epidemiology

KW - group A streptococcus

KW - sudden death

UR - https://www.scopus.com/pages/publications/105003000124

U2 - 10.1097/INF.0000000000004775

DO - 10.1097/INF.0000000000004775

M3 - Article

C2 - 40063740

SN - 0891-3668

VL - 44

SP - e156-e160

JO - Pediatric infectious disease journal

JF - Pediatric infectious disease journal

IS - 5

ER -

Group A Streptococcal Disease in Sudden Unexpected Death in Youth in the Pre- and Post-COVID-19 Era

Abstract

UN SDGs

Keywords

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