Article, 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, ISSN 1095-9157, 0896-8411, Volume 147, Page 103265, 10.1016/j.jaut.2024.103265

Contributors

Caorsi, Roberta [1] Consolaro, Alessandro [1] [2] Speziani, Camilla [1] Sözeri, Betül Y 0000-0002-5079-5644 [3] Ulu, Kadir 0000-0002-8197-6077 [3] Faugier-Fuentes, Enrique 0000-0001-7807-1296 [4] Menchaca-Aguayo, Héctor F 0000-0002-8317-9506 [4] Ozen, Seza [5] Sener, Seher 0000-0003-1564-8996 [5] Rahman, Shahana Akhter [6] Islam, Mohammad Imnul [6] Haerynck, Filomeen 0000-0001-9161-7361 [7] Simonini, Gabriele 0000-0002-5919-4861 [8] Mastri, Mariel Viviana [9] Avcin, Tadej [10] Srsen, Sasa 0000-0001-6555-2613 [11] De Albuquerque Pedrosa Fernandes, Taciana 0000-0001-7681-8915 [12] Stanevicha, Valda [13] Vojinovic, Jelena 0000-0002-6701-3370 [14] Sobh, Ali 0000-0001-7047-076X [15] Fingerhutova, Sarka [16] Minxová, Lenka [17] Gagro, Alenka 0000-0001-6308-6460 [18] Fonseca, Adriana Rodrigues 0000-0002-3536-3552 [19] Pandya, Devang [20] Varbanova, Boriana [21] Sánchez-Manubens, Judith 0000-0002-7615-4765 [22] Ganeva, Margarita [23] Montin, Davide [24] Boyarchuk, Okana [25] Minghini, Andrea [1] Bracaglia, Claudia [26] Brogan, Paul A 0000-0001-6178-6893 [27] Candotti, Fabio Magalhaes 0000-0001-6399-6042 [28] Cattalini, Marco [29] Meyts, Isabelle [30] Minoia, Francesca 0000-0002-5093-8422 [31] Taddio, Andrea [32] Wouters, Carine Helena 0000-0002-6426-8845 [30] [33] De Benedetti, Fabrizio [26] Bovis, Francesca [2] Ravelli, Angelo [1] Ruperto, Nicolino [1] Gattorno, Marco (Corresponding author) [1] group, HyperPED-COVID study Immunodeficiency, Autoinflammatory and Autoimmune Diseases) ERN-RITA (European Reference Network for Rare ESID ISSAID and the Organisation, Paediatric Rheumatology International Trials Bilginer, Yelda [34] Laila, Kamrul [6] Islam, Mohammed Mahbubul [6] Meertens, Bram [7] Hoste, Levi 0000-0001-9733-1049 [7] Dehoorne, Joke [7] Schelstraete, Petra [7] Vandekerckhove, Kristof D H M 0000-0002-1481-0878 [7] Willems, Jef 0000-0001-8250-8964 [7] Matthijs, Inge [35] Beretta, Giovanni Filocamo e Gisella Beatrice [31] Magalhães, Claudia Saad 0000-0002-7631-7093 [12] Chubata, Oksana [36] Ricci, Francesca [29] Vukovic, Antonija [11] Temelkova, Katya [37] Avramovič, Mojca Zajc 0000-0003-1680-628X [10] Emeršič, Nina 0009-0001-5975-3969 [10] Bizjak, Masa [10] Vesel, Tina Tajnšek [10] Rodrigues, Marta Felix [19] De Almeida, Rozana Gasparello [19] Lukjanovica, Kristine [38] Elnagdy, Marwa H 0000-0002-5542-1684 [39] Soliman, Ahmed 0000-0003-3964-3778 [39] Terifajova, Eva [17] Brejchova, Ivana [40] Magner, Martin [41] Myrup, Charlotte 0000-0001-6960-7466 [42] Vougiouka, Olga [43] Jelusic, Marija 0000-0002-1728-4260 [44] La Torre, Francesco [45] Rigante, Donato [46] Maggio, Maria Cristina [47] Verdoni, Lucio [48] Rubio-Pérez, Nadina Eugenia 0000-0002-7685-3734 [49] Cornejo, Gabriel Vega 0000-0001-7637-2933 [50] Treviño, Ana Victoria Villarreal 0000-0002-6758-2450 [49] Brito, Iva [51] Oliveira-Ramos, Filipa 0000-0001-7132-8909 [52] Alexeeva, Ekaterina I 0000-0002-7297-2947 [53] Chasnyk, Vyacheslav [54] Arkachaisri, Thaschawee 0000-0002-1387-1169 [55] Boyko, Yaryna 0000-0001-6264-0917 [56] Vyzhga, Yulia 0000-0002-3237-9962 [57] Samsonenko, Svitlana [58]

Affiliations

  1. [1] Istituto Giannina Gaslini
  2. [NORA names: Italy; Europe, EU; OECD];
  3. [2] University of Genoa
  4. [NORA names: Italy; Europe, EU; OECD];
  5. [3] Ümraniye Eğitim ve Araştırma Hastanesi
  6. [NORA names: Turkey; Asia, Middle East; OECD];
  7. [4] Hospital Infantil de México Federico Gómez
  8. [NORA names: Mexico; America, Central; OECD];
  9. [5] Hacettepe University
  10. [NORA names: Turkey; Asia, Middle East; OECD];

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

America, Central-Eastern, Central-Eastern Europe, Europe, European countries, International Society, LRC, Latin, Latin America, MIS-C, Western, Western Europe, admission, age, age groups, analysis, anti-inflammatory treatment, area, associated with unfavorable outcomes, biological drugs, biology, cardiac involvement, care facilities, center, children, clinical outcomes, clinical presentation, correlation, countries, data, delay, disease, disease presentation, disorders, distribution, drug, facilities, factors, geographical areas, geographical distribution, geographical macro-areas, group, heparin prophylaxis, hospital, hospital admission, hypertension, immunological disorders, impact, impact of age, inflammatory syndrome, intensity, intensive care facilities, international multicenter registry, international registry, involvement, lymphopenia, macro-areas, mechanical ventilation, mortality, mortality rate, multicenter, multicenter registry, multicenter retrospective study, multisystem, multisystem inflammatory syndrome, multivariate analysis, non-users, older children, older patients, outcomes, patients, presence, presentation, prophylaxis, rate, referral, registry, relationship, renal hypertension, residents, resource-limited countries, respiratory support, retrospective study, sequelae, sequelae rate, severe cardiac involvement, severe outcomes, society, stratification, stratification of patients, study, support, syndrome, time, time to referral, treatment, unfavorable outcome, ventilation, young children

Funders

  • Capital Region of Denmark
  • European Commission

Data Provider: Digital Science