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Meta-Analysis
. 2021 Oct;37(10):1619-1628.
doi: 10.1016/j.cjca.2021.05.014. Epub 2021 Jun 6.

Kawasaki Disease Shock Syndrome vs Classical Kawasaki Disease: A Meta-analysis and Comparison With SARS-CoV-2 Multisystem Inflammatory Syndrome

Loubna Lamrani  1 , Cedric Manlhiot  2 , Matthew D Elias  3 , Nadine F Choueiter  4 , Audrey Dionne  5 , Ashraf S Harahsheh  6 , Michael A Portman  7 , Brian W McCrindle  8 , Nagib Dahdah  9
Affiliations
Meta-Analysis

Kawasaki Disease Shock Syndrome vs Classical Kawasaki Disease: A Meta-analysis and Comparison With SARS-CoV-2 Multisystem Inflammatory Syndrome

Loubna Lamrani et al. Can J Cardiol. 2021 Oct.

Abstract
in English, French

Background: The emergence of increasing reports worldwide of a severe inflammatory process and shock in pediatric patients resembling Kawasaki disease (KD)-and, more specifically, Kawasaki disease shock syndrome (KDSS)-prompted us to explore KDSS in a preamble of a systematic comparison between the 2 conditions.

Methods: We completed a systematic review of KDSS and performed a meta-analysis comparison between reported KDSS cases and KD controls.

Results: A total of 10 case-control series were included in the meta-analysis. Patients with KDSS were older (38.4 ± 30.6 vs 21.9 ± 19.5 months; P < 0.001) compared with standard KD with equal sex distribution and completeness of clinical diagnostic criteria. KDSS present higher C-reactive protein (59.4 ± 29.2 mg/dL vs 20.8 ± 14.8 mg/dL; P < 0.001), lower albumin (2.7 ± 0.5 g/dL vs 3.3 ± 0.5 g/dL; P < 0.01), and lower platelets (255 ± 149 109/L vs 394 ± 132 109/L; P < 0.001) but only borderline higher white blood cells (P = 0.06). Differences in alanine transaminase, aspartate aminotransferase, and erythrocyte sedimentation rate were nonsignificant. The odds of intravenous immunoglobulin resistance (44.4% vs 9.6%; (P < 0.001) and the hospital length of stay (10.9 ± 5.8 vs 5.0 ± 3.0 days; P < 0.001) were higher in KDSS, as were the odds of coronary-artery abnormalities (33.9% vs 8.6%; P < 0.001).

Conclusions: This first meta-analysis on KDSS vs KD represents a basis for future works on KDSS and opens the opportunity for future multicentre studies in the search of causal relationships between presenting elements and the eventual complications of KDSS. The similarities between SARS-CoV-2 multisystem inflammatory syndrome in children and KDSS open new horizons to the understanding of the etiology and pathophysiology related to KDSS.

Contexte: À l’échelle mondiale, de plus en plus d'enfants sont touchés par un processus inflammatoire grave avec état de choc mimant la maladie de Kawasaki – nommément le syndrome de choc de la maladie de Kawasaki (Kawasaki disease shock syndrome ou KDSS). Cette situation nous a incités à mieux caractériser le KDSS en préambule à une comparaison systématique des deux pathologies.

Méthodologie: Nous avons passé systématiquement en revue les cas de KDSS et effectué une méta-analyse comparant les cas déclarés de KDSS à des témoins atteints de la maladie de Kawasaki.

Résultats: La méta-analyse a porté sur 10 séries cas/témoins en tout. Les patients atteints de KDSS étaient plus âgés (38,4 ± 30,6 mois vs 21,9 ± 19,5 mois; p < 0,001) que les patients présentant la forme classique de la maladie de Kawasaki, il y avait répartition égale par sexe et sur le taux des critères de diagnostic clinique réunis. Le KDSS se caractérise par une concentration plus élevée de protéine C-réactive (59,4 ± 29,2 mg/dl vs 20,8 ± 14,8 mg/dl; p < 0,001), une concentration plus faible d'albumine (2,7 ± 0,5 g/dl vs 3,3 ± 0,5 g/dl; p < 0,01) et un nombre moins élevé de plaquettes (255 ± 149 109/L vs 394 ± 132 109/L; p < 0,001), mais les taux de globules blancs sont à peine plus élevés (p = 0,06). Les différences relatives à l'alanine transaminase, à l'aspartate aminotransférase et à la vitesse de sédimentation érythrocytaire n’étaient pas significatives. Le risque de résistance aux immunoglobulines intraveineuses (44,4 % vs 9,6 %; (p < 0,001) et la durée d'hospitalisation (10,9 ± 5,8 vs 5,0 ± 3,0 jours; p < 0,001) étaient plus grands chez les patients atteints de KDSS, tout comme le risque d'anomalies coronariennes (33,9 % vs 8,6 %; p < 0,001).

Conclusions: Cette première méta-analyse comparative portant sur le KDSS et la maladie de Kawasaki jette les bases de travaux ultérieurs sur le KDSS et prépare la voie pour des études multicentriques axées sur la recherche des relations causales entre les signes cliniques et les complications possibles du KDSS. Les similitudes entre le syndrome inflammatoire multisystémique pédiatrique lié au SRAS-CoV-2 et le KDSS ouvrent de nouvelles perspectives en matière de compréhension des aspects étiologiques et physiopathologiques du KDSS.

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Figures

Figure 1
Figure 1
Breakdown of systematic review and meta-analysis. Orange frames: total cases included in the meta-analysis; green frames: 132 total cases included in the aggregated data. KD, Kawasaki disease; KDSS, Kawasaki disease shock syndrome.
Figure 2
Figure 2
Meta-regression results reporting clinical aspects of Kawasaki disease shock syndrome/Kawasaki disease(observed outcome) by incorporated study and by combined effect representing significant factors (P values for combined effect). IVIG, intravenous immunoglobulin; RE, random-effect.
Figure 3
Figure 3
Meta-regression results reporting laboratory data of Kawasaki disease shock syndrome/Kawasaki disease (observed outcome) of Kawasaki disease shock syndrome by incorporated study and by combined effect representing significant factors (P values for combined effect). RE, random-effect.
See this image and copyright information in PMC

References

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MeSH terms

Supplementary concepts