The HyperPed-COVID international registry: Impact of age of onset, disease presentation and geographical distribution on the final outcome of MIS-C

Roberta Caorsi; Alessandro Consolaro; Camilla Speziani; Betul Sozeri; Kadir Ulu; Enrique Faugier-Fuentes; Hector Menchaca-Aguayo; Seza Ozen; Seher Sener; Shahana Akhter Rahman; Mohammad Imnul Islam; Filomeen Haerynck; Gabriele Simonini; Mariel Viviana Mastri; Tadej Avcin; Saša Sršen; Taciana de Albuquerque Pedrosa Fernandes; Valda Stanevicha; Jelena Vojinovic; Ali Sobh; Sarka Fingerhutova; Lenka Minxova; Alenka Gagro; Adriana Rodrigues Fonseca; Devang Pandya; Boriana Varbanova; Judith Sánchez-Manubens; Margarita Ganeva; Davide Montin; Okana Boyarchuk; Andrea Minghini; Claudia Bracaglia; Paul Brogan; Fabio Candotti; Marco Cattalini; Isabelle Meyts; Francesca Minoia; Andrea Taddio; Carine Wouters; Fabrizio De Benedetti; Francesca Bovis; Angelo Ravelli; Nicolino Ruperto; Marco Gattorno; Yelda Bilginer; Kamrul Laila; Mohammed Mahbubul Islam; Bram Meertens; Levi Hoste; Charlotte Myrup; HyperPED-COVID study group

doi:10.1016/j.jaut.2024.103265

The HyperPed-COVID international registry: Impact of age of onset, disease presentation and geographical distribution on the final outcome of MIS-C

Roberta Caorsi, Alessandro Consolaro, Camilla Speziani, Betul Sozeri, Kadir Ulu, Enrique Faugier-Fuentes, Hector Menchaca-Aguayo, Seza Ozen, Seher Sener, Shahana Akhter Rahman, Mohammad Imnul Islam, Filomeen Haerynck, Gabriele Simonini, Mariel Viviana Mastri, Tadej Avcin, Saša Sršen, Taciana de Albuquerque Pedrosa Fernandes, Valda Stanevicha, Jelena Vojinovic, Ali SobhSarka Fingerhutova, Lenka Minxova, Alenka Gagro, Adriana Rodrigues Fonseca, Devang Pandya, Boriana Varbanova, Judith Sánchez-Manubens, Margarita Ganeva, Davide Montin, Okana Boyarchuk, Andrea Minghini, Claudia Bracaglia, Paul Brogan, Fabio Candotti, Marco Cattalini, Isabelle Meyts, Francesca Minoia, Andrea Taddio, Carine Wouters, Fabrizio De Benedetti, Francesca Bovis, Angelo Ravelli, Nicolino Ruperto, Marco Gattorno, Yelda Bilginer, Kamrul Laila, Mohammed Mahbubul Islam, Bram Meertens, Levi Hoste, Charlotte Myrup, HyperPED-COVID study group

Department of Paediatrics and Adolescent Medicine

8 Citations (Scopus)

Abstract

OBJECTIVES: The aim of the study was to establish an international multicenter registry to collect data on patients with Multisystem Inflammatory Syndrome in Children (MIS-C), in order to highlight a relationship between clinical presentation, age of onset and geographical distribution on the clinical outcome.

STUDY DESIGN: Multicenter retrospective study involving different international societies for rare immunological disorders.1009 patients diagnosed with MIS-C between March and September 2022, from 48 centers and 22 countries were collected. Five age groups (<1, 1-4, 5-11, 12-16, >16 years) and four geographic macro-areas, Western Europe, Central-Eastern Europe, Latin America, Asian-African resource-limited countries (LRC), were identified.

RESULTS: Time to referral was significantly higher in LRC. Intensive anti-inflammatory treatment, including biologics, respiratory support and mechanic ventilation were more frequently used in older children and in European countries. The mortality rate was higher in very young children (<1 year), in older patients (>16 years of age) and in LRC. Multivariate analysis identified the residence in LRC, presence of severe cardiac involvement, renal hypertension, lymphopenia and non-use of heparin prophylaxis, as the factors most strongly associated with unfavorable outcomes.

CONCLUSIONS: The stratification of patients by age and geographic macro-area provided insights into the clinical presentation, treatment and outcome of MIS-C. The mortality and sequelae rates exhibited a correlation with the age and geographical areas. Patients admitted and treated in LRC displayed more severe outcomes, possibly due to delays in hospital admission and limited access to biologic drugs and to intensive care facilities.

Original language	English
Article number	103265
Journal	Journal of Autoimmunity
Volume	147
ISSN	0896-8411
DOIs	https://doi.org/10.1016/j.jaut.2024.103265
Publication status	Published - 2024

Keywords

bDMARDs
Disease outcome
Limited resources countries
MIS-C
Registry

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10.1016/j.jaut.2024.103265

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Caorsi, R., Consolaro, A., Speziani, C., Sozeri, B., Ulu, K., Faugier-Fuentes, E., Menchaca-Aguayo, H., Ozen, S., Sener, S., Akhter Rahman, S., Imnul Islam, M., Haerynck, F., Simonini, G., Mastri, M. V., Avcin, T., Sršen, S., de Albuquerque Pedrosa Fernandes, T., Stanevicha, V., Vojinovic, J., ... HyperPED-COVID study group (2024). The HyperPed-COVID international registry: Impact of age of onset, disease presentation and geographical distribution on the final outcome of MIS-C. Journal of Autoimmunity, 147, Article 103265. https://doi.org/10.1016/j.jaut.2024.103265

Caorsi, R, Consolaro, A, Speziani, C, Sozeri, B, Ulu, K, Faugier-Fuentes, E, Menchaca-Aguayo, H, Ozen, S, Sener, S, Akhter Rahman, S, Imnul Islam, M, Haerynck, F, Simonini, G, Mastri, MV, Avcin, T, Sršen, S, de Albuquerque Pedrosa Fernandes, T, Stanevicha, V, Vojinovic, J, Sobh, A, Fingerhutova, S, Minxova, L, Gagro, A, Rodrigues Fonseca, A, Pandya, D, Varbanova, B, Sánchez-Manubens, J, Ganeva, M, Montin, D, Boyarchuk, O, Minghini, A, Bracaglia, C, Brogan, P, Candotti, F, Cattalini, M, Meyts, I, Minoia, F, Taddio, A, Wouters, C, De Benedetti, F, Bovis, F, Ravelli, A, Ruperto, N, Gattorno, M, Bilginer, Y, Laila, K, Islam, MM, Meertens, B, Hoste, L, Myrup, C & HyperPED-COVID study group 2024, 'The HyperPed-COVID international registry: Impact of age of onset, disease presentation and geographical distribution on the final outcome of MIS-C', Journal of Autoimmunity, vol. 147, 103265. https://doi.org/10.1016/j.jaut.2024.103265

@article{de41c70c8d1e4e6f9b2f02f3735ed223,

title = "The HyperPed-COVID international registry: Impact of age of onset, disease presentation and geographical distribution on the final outcome of MIS-C",

abstract = "OBJECTIVES: The aim of the study was to establish an international multicenter registry to collect data on patients with Multisystem Inflammatory Syndrome in Children (MIS-C), in order to highlight a relationship between clinical presentation, age of onset and geographical distribution on the clinical outcome.STUDY DESIGN: Multicenter retrospective study involving different international societies for rare immunological disorders.1009 patients diagnosed with MIS-C between March and September 2022, from 48 centers and 22 countries were collected. Five age groups (<1, 1-4, 5-11, 12-16, >16 years) and four geographic macro-areas, Western Europe, Central-Eastern Europe, Latin America, Asian-African resource-limited countries (LRC), were identified.RESULTS: Time to referral was significantly higher in LRC. Intensive anti-inflammatory treatment, including biologics, respiratory support and mechanic ventilation were more frequently used in older children and in European countries. The mortality rate was higher in very young children (<1 year), in older patients (>16 years of age) and in LRC. Multivariate analysis identified the residence in LRC, presence of severe cardiac involvement, renal hypertension, lymphopenia and non-use of heparin prophylaxis, as the factors most strongly associated with unfavorable outcomes.CONCLUSIONS: The stratification of patients by age and geographic macro-area provided insights into the clinical presentation, treatment and outcome of MIS-C. The mortality and sequelae rates exhibited a correlation with the age and geographical areas. Patients admitted and treated in LRC displayed more severe outcomes, possibly due to delays in hospital admission and limited access to biologic drugs and to intensive care facilities.",

keywords = "bDMARDs, Disease outcome, Limited resources countries, MIS-C, Registry",

author = "Roberta Caorsi and Alessandro Consolaro and Camilla Speziani and Betul Sozeri and Kadir Ulu and Enrique Faugier-Fuentes and Hector Menchaca-Aguayo and Seza Ozen and Seher Sener and \{Akhter Rahman\}, Shahana and \{Imnul Islam\}, Mohammad and Filomeen Haerynck and Gabriele Simonini and Mastri, \{Mariel Viviana\} and Tadej Avcin and Sa{\v s}a Sr{\v s}en and \{de Albuquerque Pedrosa Fernandes\}, Taciana and Valda Stanevicha and Jelena Vojinovic and Ali Sobh and Sarka Fingerhutova and Lenka Minxova and Alenka Gagro and \{Rodrigues Fonseca\}, Adriana and Devang Pandya and Boriana Varbanova and Judith S{\'a}nchez-Manubens and Margarita Ganeva and Davide Montin and Okana Boyarchuk and Andrea Minghini and Claudia Bracaglia and Paul Brogan and Fabio Candotti and Marco Cattalini and Isabelle Meyts and Francesca Minoia and Andrea Taddio and Carine Wouters and \{De Benedetti\}, Fabrizio and Francesca Bovis and Angelo Ravelli and Nicolino Ruperto and Marco Gattorno and Yelda Bilginer and Kamrul Laila and Islam, \{Mohammed Mahbubul\} and Bram Meertens and Levi Hoste and Charlotte Myrup and \{HyperPED-COVID study group\}",

year = "2024",

doi = "10.1016/j.jaut.2024.103265",

language = "English",

volume = "147",

journal = "Journal of Autoimmunity",

issn = "0896-8411",

publisher = "Academic Press",

}

TY - JOUR

T1 - The HyperPed-COVID international registry

T2 - Impact of age of onset, disease presentation and geographical distribution on the final outcome of MIS-C

AU - Caorsi, Roberta

AU - Consolaro, Alessandro

AU - Speziani, Camilla

AU - Sozeri, Betul

AU - Ulu, Kadir

AU - Faugier-Fuentes, Enrique

AU - Menchaca-Aguayo, Hector

AU - Ozen, Seza

AU - Sener, Seher

AU - Akhter Rahman, Shahana

AU - Imnul Islam, Mohammad

AU - Haerynck, Filomeen

AU - Simonini, Gabriele

AU - Mastri, Mariel Viviana

AU - Avcin, Tadej

AU - Sršen, Saša

AU - de Albuquerque Pedrosa Fernandes, Taciana

AU - Stanevicha, Valda

AU - Vojinovic, Jelena

AU - Sobh, Ali

AU - Fingerhutova, Sarka

AU - Minxova, Lenka

AU - Gagro, Alenka

AU - Rodrigues Fonseca, Adriana

AU - Pandya, Devang

AU - Varbanova, Boriana

AU - Sánchez-Manubens, Judith

AU - Ganeva, Margarita

AU - Montin, Davide

AU - Boyarchuk, Okana

AU - Minghini, Andrea

AU - Bracaglia, Claudia

AU - Brogan, Paul

AU - Candotti, Fabio

AU - Cattalini, Marco

AU - Meyts, Isabelle

AU - Minoia, Francesca

AU - Taddio, Andrea

AU - Wouters, Carine

AU - De Benedetti, Fabrizio

AU - Bovis, Francesca

AU - Ravelli, Angelo

AU - Ruperto, Nicolino

AU - Gattorno, Marco

AU - Bilginer, Yelda

AU - Laila, Kamrul

AU - Islam, Mohammed Mahbubul

AU - Meertens, Bram

AU - Hoste, Levi

AU - Myrup, Charlotte

AU - HyperPED-COVID study group

PY - 2024

Y1 - 2024

N2 - OBJECTIVES: The aim of the study was to establish an international multicenter registry to collect data on patients with Multisystem Inflammatory Syndrome in Children (MIS-C), in order to highlight a relationship between clinical presentation, age of onset and geographical distribution on the clinical outcome.STUDY DESIGN: Multicenter retrospective study involving different international societies for rare immunological disorders.1009 patients diagnosed with MIS-C between March and September 2022, from 48 centers and 22 countries were collected. Five age groups (<1, 1-4, 5-11, 12-16, >16 years) and four geographic macro-areas, Western Europe, Central-Eastern Europe, Latin America, Asian-African resource-limited countries (LRC), were identified.RESULTS: Time to referral was significantly higher in LRC. Intensive anti-inflammatory treatment, including biologics, respiratory support and mechanic ventilation were more frequently used in older children and in European countries. The mortality rate was higher in very young children (<1 year), in older patients (>16 years of age) and in LRC. Multivariate analysis identified the residence in LRC, presence of severe cardiac involvement, renal hypertension, lymphopenia and non-use of heparin prophylaxis, as the factors most strongly associated with unfavorable outcomes.CONCLUSIONS: The stratification of patients by age and geographic macro-area provided insights into the clinical presentation, treatment and outcome of MIS-C. The mortality and sequelae rates exhibited a correlation with the age and geographical areas. Patients admitted and treated in LRC displayed more severe outcomes, possibly due to delays in hospital admission and limited access to biologic drugs and to intensive care facilities.

AB - OBJECTIVES: The aim of the study was to establish an international multicenter registry to collect data on patients with Multisystem Inflammatory Syndrome in Children (MIS-C), in order to highlight a relationship between clinical presentation, age of onset and geographical distribution on the clinical outcome.STUDY DESIGN: Multicenter retrospective study involving different international societies for rare immunological disorders.1009 patients diagnosed with MIS-C between March and September 2022, from 48 centers and 22 countries were collected. Five age groups (<1, 1-4, 5-11, 12-16, >16 years) and four geographic macro-areas, Western Europe, Central-Eastern Europe, Latin America, Asian-African resource-limited countries (LRC), were identified.RESULTS: Time to referral was significantly higher in LRC. Intensive anti-inflammatory treatment, including biologics, respiratory support and mechanic ventilation were more frequently used in older children and in European countries. The mortality rate was higher in very young children (<1 year), in older patients (>16 years of age) and in LRC. Multivariate analysis identified the residence in LRC, presence of severe cardiac involvement, renal hypertension, lymphopenia and non-use of heparin prophylaxis, as the factors most strongly associated with unfavorable outcomes.CONCLUSIONS: The stratification of patients by age and geographic macro-area provided insights into the clinical presentation, treatment and outcome of MIS-C. The mortality and sequelae rates exhibited a correlation with the age and geographical areas. Patients admitted and treated in LRC displayed more severe outcomes, possibly due to delays in hospital admission and limited access to biologic drugs and to intensive care facilities.

KW - bDMARDs

KW - Disease outcome

KW - Limited resources countries

KW - MIS-C

KW - Registry

UR - https://www.scopus.com/pages/publications/85194895809

U2 - 10.1016/j.jaut.2024.103265

DO - 10.1016/j.jaut.2024.103265

M3 - Journal article

C2 - 38838452

SN - 0896-8411

VL - 147

JO - Journal of Autoimmunity

JF - Journal of Autoimmunity

M1 - 103265

ER -

The HyperPed-COVID international registry: Impact of age of onset, disease presentation and geographical distribution on the final outcome of MIS-C

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Keywords

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