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Children
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Advances in Pediatric Multisystem Inflammatory Syndrome
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Advances in Pediatric Multisystem Inflammatory Syndrome

A special issue of Children (ISSN 2227-9067).

Deadline for manuscript submissions: closed (15 January 2024) | Viewed by 4009

Special Issue Editor

Dr. Tamás Constantin

E-Mail Website
Guest Editor
2nd Department of Paediatrics, Faculty of Medicine, Semmelweis University, 1083 Budapest, Hungary
Interests: pediatric rheumatology

Special Issue Information

Dear Colleagues,

At first, after reviewing the reports coming out of China in early 2020, it seemed that hyperinflammation was one of the primary reasons for the high mortality rate of severe COVID-19 disease. The first prediction of its immunopathology came not by chance but from pediatric rheumatologists with great expertise in treating multisystem inflammatory syndromes (MIS). Dreaded representatives of these syndromes, hemophagocytic lymphohistiocytosis (HLH) and macrophage activation syndrome (MAS), are potentially life-threatening conditions.  The latter can occur in children with certain autoimmune disorders. In the last decade, pediatric rheumatology and the understanding of autoinflammation and diseases related to innate immunity have progressed dramatically. Autoinflammatory diseases are commonly associated with symptoms of hyperinflammation, while the presence of autoinflammation is often linked to causes of hyperinflammatory syndromes. Although we are certain that in severe COVID-19, we face a localized cytokine "flooding" instead of a systemic “storm,” more attention than ever before is being directed to multisystemic inflammatory syndromes.

In April 2020, United Kingdom health officials reported the first cases of what would become known as multisystem inflammatory syndrome in children (MIS-C). The children affected had various symptoms that emerged suddenly and then progressed rapidly. As word of the syndrome spread worldwide, more cases began to be reported, prompting widespread interest among pediatricians.

The latest immunological research has been translated into everyday clinical practice at lightning speed. This is, on the one hand, a fantastic achievement of medicine. On the other, general pediatricians are struggling to keep up with the many newly introduced concepts. This situation has been further complicated because clinicians, immunologists, and biologists often use overlapping but slightly different terminology.

This Special Issue aims to help readers understand the various terms for inflammation, including hyperinflammation, multisystem inflammatory syndrome, autoimmunity, and autoinflammation.

The Special Issue is currently soliciting papers that present the latest scientific results in a practical manner. We are looking for articles that not only describe the findings of scientific studies but also provide insight into how these findings can be applied in everyday life. We believe that this type of translation is essential for making scientific progress accessible to a broader audience. In addition, we feel that this approach will help to promote public understanding and support for scientific research. We encourage scientists from all disciplines to submit their work for consideration.

Dr. Tamás Constantin
Guest Editor

Manuscript Submission Information

Manuscripts should be submitted online at www.mdpi.com by registering and logging in to this website. Once you are registered, click here to go to the submission form. Manuscripts can be submitted until the deadline. All submissions that pass pre-check are peer-reviewed. Accepted papers will be published continuously in the journal (as soon as accepted) and will be listed together on the special issue website. Research articles, review articles as well as short communications are invited. For planned papers, a title and short abstract (about 100 words) can be sent to the Editorial Office for announcement on this website.

Submitted manuscripts should not have been published previously, nor be under consideration for publication elsewhere (except conference proceedings papers). All manuscripts are thoroughly refereed through a single-blind peer-review process. A guide for authors and other relevant information for submission of manuscripts is available on the Instructions for Authors page. Children is an international peer-reviewed open access monthly journal published by MDPI.

Please visit the Instructions for Authors page before submitting a manuscript. The Article Processing Charge (APC) for publication in this open access journal is 2400 CHF (Swiss Francs). Submitted papers should be well formatted and use good English. Authors may use MDPI's English editing service prior to publication or during author revisions.

Keywords

  • multisystem inflammatory syndromes
  • hyperinflammation
  • multisystem inflammatory syndrome in children
  • autoimmunity
  • autoinflammation

Published Papers (3 papers)

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18 pages, 2146 KiB  
Article
Longitudinal Characterization of Immune Response in a Cohort of Children Hospitalized with Multisystem Inflammatory Syndrome
by Laura Dotta, Daniele Moratto, Marco Cattalini, Sara Brambilla, Viviana Giustini, Antonella Meini, Maria Federica Girelli, Manuela Cortesi, Silviana Timpano, Anna Galvagni, Anna Viola, Beatrice Crotti, Alessandra Manerba, Giorgia Pierelli, Giulia Verzura, Federico Serana, Duilio Brugnoni, Emirena Garrafa, Francesca Ricci, Cesare Tomasi, Marco Chiarini and Raffaele Badolatoadd Show full author list remove Hide full author list
Children 2023, 10(6), 1069; https://doi.org/10.3390/children10061069 - 16 Jun 2023
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Abstract
Background: Multisystem Inflammatory Syndrome in Children (MIS-C) is a severe complication of SARS-CoV-2 infection caused by hyperactivation of the immune system. Methods: this is a retrospective analysis of clinical data, biochemical parameters, and immune cell subsets in 40 MIS-C patients from hospital admission [...] Read more.
Background: Multisystem Inflammatory Syndrome in Children (MIS-C) is a severe complication of SARS-CoV-2 infection caused by hyperactivation of the immune system. Methods: this is a retrospective analysis of clinical data, biochemical parameters, and immune cell subsets in 40 MIS-C patients from hospital admission to outpatient long-term follow-up. Results: MIS-C patients had elevated inflammatory markers, associated with T- and NK-cell lymphopenia, a profound depletion of dendritic cells, and altered monocyte phenotype at disease onset, while the subacute phase of the disease was characterized by a significant increase in T- and B-cell counts and a rapid decline in activated T cells and terminally differentiated B cells. Most of the immunological parameters returned to values close to the normal range during the remission phase (20–60 days after hospital admission). Nevertheless, we observed a significantly reduced ratio between recently generated and more differentiated CD8+ T- and B-cell subsets, which partially settled at longer-term follow-up determinations. Conclusions: The characterization of lymphocyte distribution in different phases of MIS-C may help to understand the course of diseases that are associated with dysregulated immune responses and to calibrate prompt and targeted treatments. Full article
(This article belongs to the Special Issue Advances in Pediatric Multisystem Inflammatory Syndrome)
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11 pages, 304 KiB  
Article
The Effect of Biologics in the Treatment of Multisystem Inflammatory Syndrome in Children (Mis-C): A Single-Center Propensity-Score-Matched Study
by Ozge Basaran, Ezgi Deniz Batu, Ummusen Kaya Akca, Erdal Atalay, Muserref Kasap Cuceoglu, Seher Sener, Zeynep Balık, Erdem Karabulut, Selman Kesici, Tevfik Karagoz, Yasemin Ozsurekci, Yelda Bilginer, Ali Bulent Cengiz and Seza Ozen
Children 2023, 10(6), 1045; https://doi.org/10.3390/children10061045 - 11 Jun 2023
Cited by 2 | Viewed by 1173
Abstract
Multisystem inflammatory syndrome in children (MIS-C) is a serious condition characterized by excessive inflammation that can arise as a complication of SARS-CoV-2 infection in children. While our understanding of COVID-19 and MIS-C has been advancing, there is still uncertainty regarding the optimal treatment [...] Read more.
Multisystem inflammatory syndrome in children (MIS-C) is a serious condition characterized by excessive inflammation that can arise as a complication of SARS-CoV-2 infection in children. While our understanding of COVID-19 and MIS-C has been advancing, there is still uncertainty regarding the optimal treatment for MIS-C. In this study, we aimed to compare the clinical and laboratory outcomes of MIS-C patients treated with IVIG plus corticosteroids (CS) to those treated with IVIG plus CS and an additional biologic drug. We used the propensity score (PS)-matching method to assess the relationships between initial treatment and outcomes. The primary outcome was a left ventricular ejection fraction of less than 55% on day 2 or beyond and/or the requirement of inotrope support on day 2 or beyond. We included 79 MIS-C patients (median age 8.51 years, 33 boys) followed in our center. Among them, 50 children (25 in each group) were allocated to the PS-matched cohort sample. The primary outcome was observed in none of the patients in the IVIG and CS group, while it occurred in eight patients in the IVIG plus CS and biologic group (p = 0.04). MIS-C is a disorder that may progress rapidly and calls for extensive care. For definitive recommendations, further studies, including randomized control trials, are required. Full article
(This article belongs to the Special Issue Advances in Pediatric Multisystem Inflammatory Syndrome)
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8 pages, 237 KiB  
Brief Report
Cytokine Storm Syndrome Triggered by Extracorporeal Membrane Oxygenation in Pediatric Patients
by Daniel D. Reiff and Randy Q. Cron
Children 2023, 10(6), 1052; https://doi.org/10.3390/children10061052 - 13 Jun 2023
Cited by 1 | Viewed by 1057
Abstract
Cytokine storm syndrome (CSS) is a serious and potentially life-threatening condition caused by severe systemic inflammation, immune activation, and a positive feedback loop of cytokine release. Typically triggered by systemic infection, malignancy, monogenic or rheumatic disease, similar patterns of hyper-inflammation have been seen [...] Read more.
Cytokine storm syndrome (CSS) is a serious and potentially life-threatening condition caused by severe systemic inflammation, immune activation, and a positive feedback loop of cytokine release. Typically triggered by systemic infection, malignancy, monogenic or rheumatic disease, similar patterns of hyper-inflammation have been seen in patients undergoing cardiopulmonary bypass (CPB) and in patients treated with extracorporeal membrane oxygenation (ECMO). Typical treatments used for the prevention and treatment of CPB/ECMO-induced hyper-inflammation have not been shown to be substantially effective. Two patients suffering from ECMO-related CSS were identified by their severe hyper-inflammatory profile and life-threatening sequelae of vasodilatory shock and respiratory failure. Anakinra, an interleukin-1 receptor antagonist, was employed as specific cytokine-directed therapy for the treatment of CSS in these two patients to good effect, with significant improvement in hyper-inflammation and cardiorespiratory status. The use of cytokine-directed therapies in CPB/ECMO-related CSS has great potential to improve the treatment and outcomes of this serious condition. Full article
(This article belongs to the Special Issue Advances in Pediatric Multisystem Inflammatory Syndrome)
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