Pediatric Inflammatory Multisystem Syndrome and Rheumatic Diseases During SARS-CoV-2 Pandemic

Schvartz, Adrien; Belot, Alexandre; Koné‐Paut, Isabelle

doi:10.3389/fped.2020.605807

Cited by 41 publications

(50 citation statements)

References 95 publications

(140 reference statements)

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“…Then, we compared these patients with patients with MIS-C from a review ( 24 ). Laboratory data were compared between our patients and patients with MIS-C in four previous papers ( 8 – 11 ). The raw data available for 10 patients with MIS-C in Italy and 6 cases of KDSS in Japan were statistically analyzed using the non-parametric Wilcoxon test.…”

Section: Methodsmentioning

confidence: 99%

“…Kawasaki disease is an acute vasculitis that occurs in childhood, and although its etiology is still unknown, it is known that many inflammatory cytokines are elevated. There have been many reports comparing KD and MIS-C, often stating that the two are partly similar but partly different ( 9 – 11 ). In particular, the overlap with KD shock syndrome (KDSS), which is KD complicated with circulatory failure and/or shock, is being discussed ( 12 , 13 ).…”

Section: Introductionmentioning

confidence: 99%

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Kawasaki Disease Shock Syndrome in Japan and Comparison With Multisystem Inflammatory Syndrome in Children in European countries

et al. 2021

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Multisystem inflammatory syndrome in children (MIS-C) is a severe Kawasaki-like illness that was first linked to severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) in European countries in the spring of 2020 and has been suggested to have overlap with Kawasaki disease shock syndrome (KDSS). There are few reports of MIS-C from Asia. This observational study aimed to identify the clinical features in children presenting with KDSS in Japan over a 5-year period and to summarize similarities and differences between KDSS and MIS-C. We retrospectively collected data on patient characteristics, clinical signs and symptoms, treatment, and prognosis including coronary artery abnormalities (CAAs), which were compared with data of patients with KDSS worldwide and patients with MIS-C from a review. KDSS was identified in 6 (1.1%) of 552 patients with Kawasaki disease (KD) treated at a single institution in Japan between 2015 and 2020 (1 in 2020). In patients with KDSS in Japan or worldwide vs. patients with MIS-C, KDSS was more likely to have a diagnosis of complete KD (100, 70 vs. 6.3%), a higher incidence of CAAs (50, 65 vs. 11%), and a greater requirement for vasoactive agonists (67, 67 vs. 43%) because of circulatory shock (100, 50 vs. 26%). Both KDSS and MIS-C had good prognosis (mortality 0, 6.7 vs. 1.7%). Although KDSS in Japan and MIS-C show some overlap in clinical symptoms, they are unlikely to be the same disease entity. KDSS is more likely to have a cardiovascular phenotype with CAAs and requires treatment with cardiovascular agents.

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Section: Methodsmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Kawasaki Disease Shock Syndrome in Japan and Comparison With Multisystem Inflammatory Syndrome in Children in European countries

et al. 2021

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“…However, autoantibodies were uniquely produced during MIS-C or KD, which supports a contribution of the humoral response to both diseases(8, 11). Finally, a role for genetic factors has been evocated in MIS-C pathogenesis as it seems to occur more frequently in children from Hispanic or African ethnicity(12, 13).…”

Section: Introductionmentioning

confidence: 99%

Superantigenic TCR Vbeta 21.3 signature in Multisystem Inflammatory Syndrome in Children

Moreews

Gouge

Bellomo

et al. 2021

Preprint

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Multiple Inflammatory Syndrome in Children (MIS-C) is the most severe pediatric form of COVID-19 and occurs in previously healthy children. MIS-C combines features of Kawasaki disease and Toxic Shock Syndrome (TSS). We characterized the immunological profile of 27 MIS-C cases in comparison with 4 KD and 4 TSS cases. Similarly to TSS, an increase of serum inflammatory cytokines (IL-6, TNF-a, CD25s) was observed in MIS-C contrasting with low expression of HLA-DR monocytes, a feature often associated with immune paralysis. Expansions of T cells expressing the Vβ21.3 T cell receptor β chain variable region were detected in both CD4 and CD8 subsets in almost 50% of patients and Vβ21.3-positive T cells expressed high level of HLA-DR highlighting their specific activation. TCR sequencing uncovered the polyclonal nature of the Vβ 21.3+ population. SARS-CoV2 antigene-specific production of interferon gamma in T cells was not increased in MIS-C T cells compared to COVID-19 patients suggesting the antigen-specific immune response in MIS-C patients is not pivotal to the manifestation. Together, these data argue in favor of a strong activation of the immune system related to a superantigenic immune response in MIS-C with a specific polyclonal Vβ21.3 expressing T cell activation.

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“…The Verdoni et al study showed that the monthly incidence of KD in an Italian province had increased 30folds (21). The significant inflammatory response of the body to the novel coronavirus alongside the epidemiological studies have been in favor of the theory that suggests COVID-19 as a trigger for the immune system and an etiology for KD's (61)(62)(63).…”

Section: Discussionmentioning

confidence: 99%

Characteristics of Children With Kawasaki Disease-Like Signs in COVID-19 Pandemic: A Systematic Review

et al. 2021

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Recent studies have shown that several children diagnosed with COVID-19 have developed Kawasaki Disease (KD)-like symptoms. This systematic review aims to assess the demographic, laboratory, and clinical characteristics of children with KD-like syndrome during the COVID-19 pandemic and evaluate efficacy of treatments and patients' outcome. A comprehensive search was carried out systematically through PubMed, Scopus, and Web of Science (WoS), medRxiv, and bioRxiv by two reviewers independently for all studies or preprints data on the demographic, laboratory, and clinical characteristics of children with K.D-like signs during the COVID-19 outbreak. Overall, 378 studies were identified by the systematic search, of which 25 studies were included in the study. The included studies involved 599 patients in total. Thirteen studies (52%) were case reports or case series, and the rest (48%) were cohort studies. In 19 studies, patients were diagnosed with Multisystem inflammatory syndrome in children (MIS-C). In 16 studies COVID-19 was diagnosed in all patients based on their polymerase chain reaction result, serological findings, and computed tomography results. Higher C-reactive protein and erythrocyte sedimentation rate level were the most prevalent laboratory findings. In most studies, patients had leucopenia with marked lymphopenia, hypoalbuminemia, and increased ferritin, as well as hyponatremia. Abnormal echocardiography and respiratory outcomes were the most common clinical outcomes. In 11 studies, all patients required intensive care unit admission. Findings of the present systematic review show that the incidence of KD-like syndrome in the COVID-19 pandemic increased significantly. Moreover, this study offers new insights in the KD-like syndrome pathogenesis and clinical spectrum during COVID-19 pandemic.

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Pediatric Inflammatory Multisystem Syndrome and Rheumatic Diseases During SARS-CoV-2 Pandemic

Cited by 41 publications

References 95 publications

Kawasaki Disease Shock Syndrome in Japan and Comparison With Multisystem Inflammatory Syndrome in Children in European countries

Kawasaki Disease Shock Syndrome in Japan and Comparison With Multisystem Inflammatory Syndrome in Children in European countries

Superantigenic TCR Vbeta 21.3 signature in Multisystem Inflammatory Syndrome in Children

Characteristics of Children With Kawasaki Disease-Like Signs in COVID-19 Pandemic: A Systematic Review

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