Predicting respiratory failure and outcome in pediatric Guillain-Barré syndrome

the Working Group GBS in Children in Germany; Austria and Switzerland and the Dutch Pediatric GBS Study Group; Joyce Roodbol; Rudolf Korinthenberg; Esmee Venema; M. C.Y. de Wit; Hester F. Lingsma; Coriene E. Catsman-Berrevoets; B. C. Jacobs; R. Korinthenberg; Joyce Roodbol; M. C.Y. de Wit; C. E. Catsman-Berrevoets; B. C. Jacobs; M. Engelen; C. E. Erasmus; C. P.W. Geleijns; I. A.W. Kotsopoulos; J. Nicolai; J. M.F. Niermeijer; E. H. Niks; J. Samijn

doi:10.1016/j.ejpn.2023.02.007

Predicting respiratory failure and outcome in pediatric Guillain-Barré syndrome

the Working Group GBS in Children in Germany, Austria and Switzerland and the Dutch Pediatric GBS Study Group, Joyce Roodbol, Rudolf Korinthenberg, Esmee Venema, M. C.Y. de Wit, Hester F. Lingsma, Coriene E. Catsman-Berrevoets, B. C. Jacobs^*, R. Korinthenberg, Joyce Roodbol, M. C.Y. de Wit, C. E. Catsman-Berrevoets, B. C. Jacobs^*, M. Engelen, C. E. Erasmus, C. P.W. Geleijns, I. A.W. Kotsopoulos, J. Nicolai, J. M.F. NiermeijerE. H. Niks, J. Samijn

^*Corresponding author for this work

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

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Abstract

Background: Guillain-Barré syndrome (GBS) has a highly variable clinical course and outcome as indicated by the risk of developing respiratory failure and residual inability to walk. Prognostic models as Erasmus GBS Respiratory Insufficiency Score (EGRIS) developed in adult patients are inaccurate in children. Our aim was to determine the prognostic factors of respiratory failure and inability to walk in children with GBS and to develop a new clinical prognostic model for individual patients (EGRIS-Kids). Methods: A multicenter retrospective cohort study was performed using the data of children (younger than 18 years) fulfilling the diagnostic criteria for GBS from the NINDS. This study was performed in two independent cohorts from centers in Germany, Switzerland, Austria (N = 265, collected 1989–2002) and The Netherlands (N = 156, collected 1987–2016). The predicted main outcomes were occurrence of respiratory failure during the disease course and inability to walk independent at one year after diagnosis. Results: In the combined cohort of 421 children, 79 (19%) required mechanical ventilation and one patient died. The EGRIS-kids was developed including: age, cranial nerve involvement and GBS disability score at admission, resulting in a 9 point score predicting risks of respiratory failure ranging from 4 to 50% (AUC = 0.71). A lower GBS disability score at nadir was the strongest predictor of recovery to independent walking (at one month: OR 0.43 95%CI 0.25–0.74). Conclusions: EGRIS-Kids and GBS disability score at admission accurately predict the risk of respiratory failure and inability to walk respectively in children with GBS, as tools to personalize the monitoring and treatment.

Original language	English
Pages (from-to)	18-24
Number of pages	7
Journal	European Journal of Paediatric Neurology
Volume	44
DOIs	https://doi.org/10.1016/j.ejpn.2023.02.007
Publication status	Published - May 2023

Bibliographical note

Funding Information:
Dr. M.C.Y. de Wit received honoraria paid to her institution by Novartis for serving on a steering committee and presenting at a conference, and has received research funding from the Epilepsiefonds (Dutch Epilepsy Foundation), Hersenstichting and Sophia Foundation.Dr. B.C. Jacobs has received funding for travel from Baxter International Inc, and has received research funding from the Netherlands Organization for Health Research and Development, Erasmus MC, Prinses Beatrix Spierfonds, Stichting Spieren voor Spieren, CSL-Behring, Grifols, Annexon, Hansa Biopharma and the GBS-CIDP Foundation International.This study was supported by a research grant from the Dutch Prinses Beatrix Spierfonds and Spieren voor Spieren (W.OR 12-04 to, J.R. M.C.Y.d W, B.C.J.). This study was possible with data provided by the Working Group GBS in Children in Germany, Austria and Switzerland and the Dutch Pediatric GBS Study Group. And all contributors to the retro- and prospective studies [6,13].

Funding Information:
This study was supported by a research grant from the Dutch Prinses Beatrix Spierfonds and Spieren voor Spieren (W.OR 12-04 to, J.R., M.C.Y.d W, B.C.J.).

Publisher Copyright:
© 2023 The Authors

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the Working Group GBS in Children in Germany, Austria and Switzerland and the Dutch Pediatric GBS Study Group, Roodbol, J., Korinthenberg, R., Venema, E., de Wit, M. C. Y., Lingsma, H. F., Catsman-Berrevoets, C. E., Jacobs, B. C., Korinthenberg, R., Roodbol, J., de Wit, M. C. Y., Catsman-Berrevoets, C. E., Jacobs, B. C., Engelen, M., Erasmus, C. E., Geleijns, C. P. W., Kotsopoulos, I. A. W., Nicolai, J., ... Samijn, J. (2023). Predicting respiratory failure and outcome in pediatric Guillain-Barré syndrome. European Journal of Paediatric Neurology, 44, 18-24. https://doi.org/10.1016/j.ejpn.2023.02.007

@article{b91687a1ebaa417b99d951a996568e1d,

title = "Predicting respiratory failure and outcome in pediatric Guillain-Barr{\'e} syndrome",

abstract = "Background: Guillain-Barr{\'e} syndrome (GBS) has a highly variable clinical course and outcome as indicated by the risk of developing respiratory failure and residual inability to walk. Prognostic models as Erasmus GBS Respiratory Insufficiency Score (EGRIS) developed in adult patients are inaccurate in children. Our aim was to determine the prognostic factors of respiratory failure and inability to walk in children with GBS and to develop a new clinical prognostic model for individual patients (EGRIS-Kids). Methods: A multicenter retrospective cohort study was performed using the data of children (younger than 18 years) fulfilling the diagnostic criteria for GBS from the NINDS. This study was performed in two independent cohorts from centers in Germany, Switzerland, Austria (N = 265, collected 1989–2002) and The Netherlands (N = 156, collected 1987–2016). The predicted main outcomes were occurrence of respiratory failure during the disease course and inability to walk independent at one year after diagnosis. Results: In the combined cohort of 421 children, 79 (19%) required mechanical ventilation and one patient died. The EGRIS-kids was developed including: age, cranial nerve involvement and GBS disability score at admission, resulting in a 9 point score predicting risks of respiratory failure ranging from 4 to 50% (AUC = 0.71). A lower GBS disability score at nadir was the strongest predictor of recovery to independent walking (at one month: OR 0.43 95%CI 0.25–0.74). Conclusions: EGRIS-Kids and GBS disability score at admission accurately predict the risk of respiratory failure and inability to walk respectively in children with GBS, as tools to personalize the monitoring and treatment.",

author = "{the Working Group GBS in Children in Germany} and {Austria and Switzerland and the Dutch Pediatric GBS Study Group} and Joyce Roodbol and Rudolf Korinthenberg and Esmee Venema and {de Wit}, {M. C.Y.} and Lingsma, {Hester F.} and Catsman-Berrevoets, {Coriene E.} and Jacobs, {B. C.} and R. Korinthenberg and Joyce Roodbol and {de Wit}, {M. C.Y.} and Catsman-Berrevoets, {C. E.} and Jacobs, {B. C.} and M. Engelen and Erasmus, {C. E.} and Geleijns, {C. P.W.} and Kotsopoulos, {I. A.W.} and J. Nicolai and Niermeijer, {J. M.F.} and Niks, {E. H.} and J. Samijn",

note = "Funding Information: Dr. M.C.Y. de Wit received honoraria paid to her institution by Novartis for serving on a steering committee and presenting at a conference, and has received research funding from the Epilepsiefonds (Dutch Epilepsy Foundation), Hersenstichting and Sophia Foundation.Dr. B.C. Jacobs has received funding for travel from Baxter International Inc, and has received research funding from the Netherlands Organization for Health Research and Development, Erasmus MC, Prinses Beatrix Spierfonds, Stichting Spieren voor Spieren, CSL-Behring, Grifols, Annexon, Hansa Biopharma and the GBS-CIDP Foundation International.This study was supported by a research grant from the Dutch Prinses Beatrix Spierfonds and Spieren voor Spieren (W.OR 12-04 to, J.R. M.C.Y.d W, B.C.J.). This study was possible with data provided by the Working Group GBS in Children in Germany, Austria and Switzerland and the Dutch Pediatric GBS Study Group. And all contributors to the retro- and prospective studies [6,13]. Funding Information: This study was supported by a research grant from the Dutch Prinses Beatrix Spierfonds and Spieren voor Spieren (W.OR 12-04 to, J.R., M.C.Y.d W, B.C.J.). Publisher Copyright: {\textcopyright} 2023 The Authors",

year = "2023",

month = may,

doi = "10.1016/j.ejpn.2023.02.007",

language = "English",

volume = "44",

pages = "18--24",

journal = "European Journal of Paediatric Neurology",

issn = "1090-3798",

}

the Working Group GBS in Children in Germany, Austria and Switzerland and the Dutch Pediatric GBS Study Group, Roodbol, J, Korinthenberg, R, Venema, E , de Wit, MCY , Lingsma, HF , Catsman-Berrevoets, CE , Jacobs, BC, Korinthenberg, R, Roodbol, J , de Wit, MCY , Catsman-Berrevoets, CE , Jacobs, BC, Engelen, M, Erasmus, CE, Geleijns, CPW, Kotsopoulos, IAW, Nicolai, J, Niermeijer, JMF, Niks, EH & Samijn, J 2023, 'Predicting respiratory failure and outcome in pediatric Guillain-Barré syndrome', European Journal of Paediatric Neurology, vol. 44, pp. 18-24. https://doi.org/10.1016/j.ejpn.2023.02.007

Predicting respiratory failure and outcome in pediatric Guillain-Barré syndrome. / the Working Group GBS in Children in Germany; Austria and Switzerland and the Dutch Pediatric GBS Study Group ; Roodbol, Joyce et al.
In: European Journal of Paediatric Neurology, Vol. 44, 05.2023, p. 18-24.

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

TY - JOUR

T1 - Predicting respiratory failure and outcome in pediatric Guillain-Barré syndrome

AU - the Working Group GBS in Children in Germany

AU - Austria and Switzerland and the Dutch Pediatric GBS Study Group

AU - Roodbol, Joyce

AU - Korinthenberg, Rudolf

AU - Venema, Esmee

AU - de Wit, M. C.Y.

AU - Lingsma, Hester F.

AU - Catsman-Berrevoets, Coriene E.

AU - Jacobs, B. C.

AU - Korinthenberg, R.

AU - Roodbol, Joyce

AU - de Wit, M. C.Y.

AU - Catsman-Berrevoets, C. E.

AU - Jacobs, B. C.

AU - Engelen, M.

AU - Erasmus, C. E.

AU - Geleijns, C. P.W.

AU - Kotsopoulos, I. A.W.

AU - Nicolai, J.

AU - Niermeijer, J. M.F.

AU - Niks, E. H.

AU - Samijn, J.

N1 - Funding Information: Dr. M.C.Y. de Wit received honoraria paid to her institution by Novartis for serving on a steering committee and presenting at a conference, and has received research funding from the Epilepsiefonds (Dutch Epilepsy Foundation), Hersenstichting and Sophia Foundation.Dr. B.C. Jacobs has received funding for travel from Baxter International Inc, and has received research funding from the Netherlands Organization for Health Research and Development, Erasmus MC, Prinses Beatrix Spierfonds, Stichting Spieren voor Spieren, CSL-Behring, Grifols, Annexon, Hansa Biopharma and the GBS-CIDP Foundation International.This study was supported by a research grant from the Dutch Prinses Beatrix Spierfonds and Spieren voor Spieren (W.OR 12-04 to, J.R. M.C.Y.d W, B.C.J.). This study was possible with data provided by the Working Group GBS in Children in Germany, Austria and Switzerland and the Dutch Pediatric GBS Study Group. And all contributors to the retro- and prospective studies [6,13]. Funding Information: This study was supported by a research grant from the Dutch Prinses Beatrix Spierfonds and Spieren voor Spieren (W.OR 12-04 to, J.R., M.C.Y.d W, B.C.J.). Publisher Copyright: © 2023 The Authors

PY - 2023/5

Y1 - 2023/5

N2 - Background: Guillain-Barré syndrome (GBS) has a highly variable clinical course and outcome as indicated by the risk of developing respiratory failure and residual inability to walk. Prognostic models as Erasmus GBS Respiratory Insufficiency Score (EGRIS) developed in adult patients are inaccurate in children. Our aim was to determine the prognostic factors of respiratory failure and inability to walk in children with GBS and to develop a new clinical prognostic model for individual patients (EGRIS-Kids). Methods: A multicenter retrospective cohort study was performed using the data of children (younger than 18 years) fulfilling the diagnostic criteria for GBS from the NINDS. This study was performed in two independent cohorts from centers in Germany, Switzerland, Austria (N = 265, collected 1989–2002) and The Netherlands (N = 156, collected 1987–2016). The predicted main outcomes were occurrence of respiratory failure during the disease course and inability to walk independent at one year after diagnosis. Results: In the combined cohort of 421 children, 79 (19%) required mechanical ventilation and one patient died. The EGRIS-kids was developed including: age, cranial nerve involvement and GBS disability score at admission, resulting in a 9 point score predicting risks of respiratory failure ranging from 4 to 50% (AUC = 0.71). A lower GBS disability score at nadir was the strongest predictor of recovery to independent walking (at one month: OR 0.43 95%CI 0.25–0.74). Conclusions: EGRIS-Kids and GBS disability score at admission accurately predict the risk of respiratory failure and inability to walk respectively in children with GBS, as tools to personalize the monitoring and treatment.

AB - Background: Guillain-Barré syndrome (GBS) has a highly variable clinical course and outcome as indicated by the risk of developing respiratory failure and residual inability to walk. Prognostic models as Erasmus GBS Respiratory Insufficiency Score (EGRIS) developed in adult patients are inaccurate in children. Our aim was to determine the prognostic factors of respiratory failure and inability to walk in children with GBS and to develop a new clinical prognostic model for individual patients (EGRIS-Kids). Methods: A multicenter retrospective cohort study was performed using the data of children (younger than 18 years) fulfilling the diagnostic criteria for GBS from the NINDS. This study was performed in two independent cohorts from centers in Germany, Switzerland, Austria (N = 265, collected 1989–2002) and The Netherlands (N = 156, collected 1987–2016). The predicted main outcomes were occurrence of respiratory failure during the disease course and inability to walk independent at one year after diagnosis. Results: In the combined cohort of 421 children, 79 (19%) required mechanical ventilation and one patient died. The EGRIS-kids was developed including: age, cranial nerve involvement and GBS disability score at admission, resulting in a 9 point score predicting risks of respiratory failure ranging from 4 to 50% (AUC = 0.71). A lower GBS disability score at nadir was the strongest predictor of recovery to independent walking (at one month: OR 0.43 95%CI 0.25–0.74). Conclusions: EGRIS-Kids and GBS disability score at admission accurately predict the risk of respiratory failure and inability to walk respectively in children with GBS, as tools to personalize the monitoring and treatment.

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U2 - 10.1016/j.ejpn.2023.02.007

DO - 10.1016/j.ejpn.2023.02.007

M3 - Article

C2 - 36931152

AN - SCOPUS:85150029927

SN - 1090-3798

VL - 44

SP - 18

EP - 24

JO - European Journal of Paediatric Neurology

JF - European Journal of Paediatric Neurology

ER -

Predicting respiratory failure and outcome in pediatric Guillain-Barré syndrome

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