Predictors and management of intravenous immunoglobulin-resistant Kawasaki disease

Song, Min Seob

doi:10.3345/kjp.2019.00150

Cited by 14 publications

(13 citation statements)

References 43 publications

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“…Current follow up drug therapy for KD children with IVIG resistance mainly includes the second dose of IVIG, methylprednisolone (MP), infliximab, interleukin 1 receptor antagonist or cyclosporin [ 3 ]. However, KD patients who fail to respond to 2 doses of IVIG present a unique challenge [ 80 ]. A meta-analysis comprising 372 refractory KD patients conducted in 2019 by Chan et al [ 81 ] revealed that infliximab, MP, and second IVIG infusion showed no significant differences in the cardioprotective effect or the rate of treatment resistance.…”

Section: Discussionmentioning

confidence: 99%

Is there an association between intravenous immunoglobulin resistance and coronary artery lesion in Kawasaki disease?—Current evidence based on a meta-analysis

Zheng

Peng

et al. 2021

PLoS ONE

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Background Coronary artery lesion (CAL) caused by Kawasaki disease (KD) is a leading cause of acquired heart disease in children. Initial treatment of intravenous immunoglobulin (IVIG) can reduce the incidence of CAL. Although most of the current studies have shown a certain correlation between CAL and IVIG resistance, the conclusions are not completely consistent. Thus, we performed this meta-analysis to evaluate the association between IVIG resistance and CAL in KD. Methods PubMed, EMBASE, the Cochrane Central Register of Controlled Trials, and China National Knowledge Infrastructure through April 21, 2020 were searched to detect relevant studies. Data analysis was performed with STATA 15.1. Results A total of 53 relevant studies were eligible to this analysis, including 30312 KD patients, of which 4750 were IVIG resistance and 25562 were responders. There was a significant difference found between IVIG resistance and IVIG response groups in the incidence of CAL (P < 0.001, odds ratio (OR), 3.89; 95% confidence interval (CI) (3.18, 4.75)). The heterogeneity test results showed that the I2 value was 74.8%. The meta-regression analysis showed that the study regions might be the sources of heterogeneity. The subgroup analysis suggested that the incidence of CAL in the IVIG resistance group was still higher than that in the IVIG response group under different regions, IVIG resistance diagnostic criteria, CAL diagnostic criteria, and study types. Meanwhile, the sensitivity analysis did not find any significant impact from every single study. Conclusions This is the first meta-analysis to reveal the incidence of CAL was associated with IVIG resistance in KD patients. Further well-designed studies with uniform criteria are needed to evaluate the incidence of CAL in IVIG resistant patients.

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Section: Discussionmentioning

confidence: 99%

Is there an association between intravenous immunoglobulin resistance and coronary artery lesion in Kawasaki disease?—Current evidence based on a meta-analysis

Zheng

Peng

et al. 2021

PLoS ONE

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“…While steroids are the first line of management in most of the immune vasculitis; its use is controversial in Kawasaki disease (11,12). Intravenous immunoglobulins is the first line of management to prevent the coronary artery disease in Kawasaki disease (13). Incomplete or atypical Kawasaki disease should be taken into consideration in case of all children with unexplained fever for more than 5 days, associated with 2 or 3 of the main clinical findings of Kawasaki disease (4).…”

Section: Discussionmentioning

confidence: 99%

“…The diagnosis of incomplete Kawasaki disease is based on presence of any three of the supplemental laboratory criteria: anaemia, leucocytosis, thrombocytosis, elevated alanine aminotransferase, hypoalbuminemia and sterile pyuria. Cardiac complications, mostly coronary artery aneurysm, can occur in 20% to 25% of untreated patients and in 4% of treated patients (13). Other aneurysms in different medium sized arteries are documented ( e.g.…”

Section: Discussionmentioning

confidence: 99%

Unusual Presentation of Atypical Kawasaki Disease in a Child with Thrombocytopenia and Spontaneously Femoral Artery Leak: A Case Report

Eid

Badr

Hamza

et al. 2021

Pediatric Sciences Journal

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Kawasaki disease (KD) is an acute systemic vascular disease that affects mostly medium sized and small vessels. The most serious of which is coronary artery disease. Vasculitis involves proliferative granulomatous inflammation and resolves by cicatrization if not treated. It is generally a self-limited disease and its highest incidence is in children under five years. The diagnostic criteria include the presence of fever for at least 5 days along with four of five other clinical features (rash, mucositis, conjunctival injection, cervical lymphadenopathy or extremity changes). Atypical Kawasaki disease includes patients who meet only 2 or 3 of the 5 criteria for diagnosis. We here report a 4-year old male patient who presented with fever, mucositis, perineal rash and evidence of de novo femoral vasculitis leading to spontaneously leaking femoral arteritis. His complete blood count revealed thrombocytopenia. The patient fulfilled the criteria for the diagnosis of atypical Kawasaki Disease.He received intravenous immunoglobulins and aspirin in anti-inflammatory doses and his condition improved. We report this unusual presentation of Kawasaki disease with spontaneous femoral artery leak, rash and thrombocytopenia. Diagnosis of atypical Kawasaki disease is very challenging but essential as initiation of specific management is life-saving. Level of Evidence of Study: IV (1).

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“…However, some severely affected patients show a persistent fever, sustained or elevated CRP and WBC, and a higher risk of CALs. For these patients, pediatricians have used various regimens including IVIG reinfusion, methylprednisolone pulse therapy (10–30 mg/kg), anti-cytokine biologics (IL-1, IL-6, and TNF-α), potent immune-suppressor such as cyclophosphamide and plasmapheresis [ 122 ]. These treatments can induce eventual defervescence but have a limited effect on preformed aneurysms and progression of CALs since the drugs cannot control the etiological substances, and ongoing inflammation of arteritis is dependent on the immune system of the patients [ 56 ].…”

Section: Treatmentmentioning

confidence: 99%

Etiological and pathophysiological enigmas of severe coronavirus disease 2019, multisystem inflammatory syndrome in children, and Kawasaki disease

2022

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During the coronavirus disease 2019 (COVID-19) pandemic, a novel multisystem inflammatory syndrome in children (MIS-C) has been reported worldwide since the first cases were reported in Europe in April 2020. MIS-C is temporally associated with severe acute respiratory syndrome coronavirus 2 infection and shows Kawasaki disease (KD)-like features. The epidemiological and clinical characteristics in COVID-19, KD, and MIS-C differ, but severe cases of each disease share similar clinical and laboratory findings such as a protracted clinical course, multiorgan involvement, and similar activated biomarkers. These findings suggest that a common control system of the host may act against severe disease insult. To solve the enigmas, we proposed the protein-homeostasis-system hypothesis in that every disease involves etiological substances and the host’s immune system controls them by their size and biochemical properties. Also, it is proposed that the etiological agents of KD and MIS-C might be certain strains in the microbiota of human species and etiological substances in severe COVID-19, KD, and MIS-C originate from pathogen-infected cells. Since disease severity depends on the amounts of inflammation-inducing substances and corresponding immune activation in the early stage of the disease, an early proper dose of corticosteroids and/or intravenous immunoglobulin (IVIG) may help reduce morbidity and possibly mortality among patients with these diseases. Corticosteroids are low cost and an analogue of host-origin cortisol among immune modulators. This study’s findings will help clinicians treating severe COVID-19, KD, and MIS-C, especially in developing countries, where IVIG and biologics supplies are insufficient.

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Predictors and management of intravenous immunoglobulin-resistant Kawasaki disease

Cited by 14 publications

References 43 publications

Is there an association between intravenous immunoglobulin resistance and coronary artery lesion in Kawasaki disease?—Current evidence based on a meta-analysis

Is there an association between intravenous immunoglobulin resistance and coronary artery lesion in Kawasaki disease?—Current evidence based on a meta-analysis

Unusual Presentation of Atypical Kawasaki Disease in a Child with Thrombocytopenia and Spontaneously Femoral Artery Leak: A Case Report

Etiological and pathophysiological enigmas of severe coronavirus disease 2019, multisystem inflammatory syndrome in children, and Kawasaki disease

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