Infliximab Treatment for Intravenous Immunoglobulin-resistant Kawasaki Disease: a Multicenter Study in Korea

Hur, Gyu Young; Song, Min Seob; Sohn, Sejung; Lee, Hyoung Doo; Kim, Jun Seok; Cho, Hwa Jin; Yoon, Kyung Lim; Joo, Chan Uhng; Hyun, Myung Chul; Kim, Chul Ho

doi:10.4070/kcj.2018.0214

Cited by 23 publications

(22 citation statements)

References 25 publications

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“…Recently, Gyu Hur (17) and Nagatomo (10) in their retrospective study observed that the early administration of infliximab may reduce the incidence of significant CAA in patients with IVIG-resistant KD.…”

Section: Discussionmentioning

confidence: 99%

Resolution of Giant Coronary Aneurisms in a Child With Refractory Kawasaki Disease Treated With Anakinra

et al. 2020

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Kawasaki disease (KD) is an acute, febrile illness of unknown etiology that mainly affects children under 5 years of age. intravenous immunoglobulin (IVIG), the standard treatment, has reduced coronary involvement to <5%. Patients who do not improve after an initial IVIG have a higher risk of developing coronary arteries aneurysms, and its optimal treatment remains controversial. We present a case of IVIG, steroids, and infliximab-resistant KD in a 9-month-old child, which developed giant aneurysms and was successfully treated with anakinra, a recombinant antagonist of the IL-1 receptor. In our case, the introduction of IL-1 receptor antagonist therapy seems to have blocked the disease from both a clinical and a laboratory point of view. We also noted a very rapid regression of coronary aneurysms passed from giant aneurysms to small ones, or, as in the case of the anterior descending artery, the complete disappearance of the aneurysm formation. We think that our case adds more evidences to the potential role of IL-1RA as therapy in some selected cases of refractory KD, in particular with severe involvement of coronary arteries, although new efficacy trials are needed to better understand the role of Anakinra in these patients.

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Section: Discussionmentioning

confidence: 99%

Resolution of Giant Coronary Aneurisms in a Child With Refractory Kawasaki Disease Treated With Anakinra

et al. 2020

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“…Several reports have been published with remarkable results when anticytokine therapy is used in the treatment of KD. A meta-analysis regarding the effect of infliximab recently published has shown that use of infliximab in IVIG refractory patients has a very strong effect on decreasing the rate of coronary aneurysms with z-scores of >5 [6], while a previous study suggested that infliximab therapy may encourage early regression of aneurysms [7]. While clinical trials of IL-1 blockade are now ongoing [5], only case reports currently exist for use of anakinra for KD.…”

Section: Discussionmentioning

confidence: 99%

Complete Regression of Giant Aneurysms in an Infant with Delayed Diagnosis and Refractory Kawasaki Disease via Combination Anticytokine Therapy: Case Report and Review of Similar Cases

Williams

Nagaraju

Gorelik

2020

Case Reports in Rheumatology

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Background. Kawasaki disease (KD) is an inflammatory vasculitis and is the most common cause of acquired childhood heart disease in developed countries. Current treatment with intravenous immunoglobulin (IVIG) is often ineffective in patients with delayed or refractory disease. We present a case of combination anticytokine therapy in an infant with delayed and refractory KD. Case Presentation. A 3-month-old infant presented with refractory KD with giant aneurysms after a delayed diagnosis of one month. Use of combination anticytokine therapy led to resolution of giant aneurysms over approximately 6 months. Conclusions. Our case is unique in effective use of anticytokine therapy in very delayed disease with giant aneurysms. Additionally, we review other cases for a broader perspective. Prospective study of anticytokine therapy for patients with giant aneurysms may be warranted.

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“…Specific blocking of TNF-α has been demonstrated to prevent coronary inflammation and the formation of CAA in mouse KD models induced by Lactobacillus casei cell wall extracts. In a previous study of a KD model in mice, it was indicated that a TNF-α anticoagulant can reduce the adhesion of neutrophils to vascular endothelial cells and the inflammation of arterial vascular endothelial cells (24,(39)(40)(41)(42). In the present meta-analysis, no significant differences in CAL, CAA, non-response rate, length of hospital stay, conjunctival injection or polymorphous exanthema between the two groups were observed; however, IFX or IFX plus IVIG significantly reduced the incidence of adverse events, the Table III.…”

Section: P-value N (Case/ ---------------------------------mentioning

confidence: 99%

Efficacy of infliximab in the treatment of Kawasaki disease: A systematic review and meta‑analysis

Lu¹,

Wang

2020

Exp Ther Med

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The present study aimed to review the relevant studies in order to determine the efficacy of infliximab (IFX) in the treatment of Kawasaki disease (KD). The relevant studies were retrieved using the PubMed, Cochrane and Embase databases. Key sources in the literature were reviewed; all articles published by July 2019 were considered for inclusion. For each study, odds ratios, mean difference and 95% confidence interval (95% CI) were assessed to evaluate study outcomes. A total of 16 studies involving 429 patients were relevant to the questions of interest of the current meta-analysis. Compared with intravenous immunoglobulin (IVIG), IFX or IFX plus IVIG significantly reduced the incidence of adverse events, including the number of patients with fever, changes in lip and oral cavity and/or cervical lymphadenopathy. The white blood cell (WBC), neutrophil and C-reactive protein (CRP) levels were also reduced in the IFX or IFX plus IVIG group compared with those in the IVIG or polyethylene glycol-treated human immunoglobulin (VGIH) groups. The platelet counts, alanine aminotransferase (ALT) levels and Z-scores were increased in the IFX or IFX plus IVIG groups compared with those in the IVIG or VGIH groups. In the single-arm studies, the incidence of coronary artery aneurysm was 0.150 (95% CI: 0.024, 0.277), the non-response rate was 0.097 (95% CI: 0.056, 0.138), and the incidence of adverse events was 0.156 (95% CI: 0.122, 0.190). IFX not only effectively reduced the incidence of fever, conjunctival injection, changes in lip and oral cavity and cervical lymphadenopathy polymorphous exanthema, but also the WBC, neutrophil, ALT and CRP levels. The platelet levels were increased in patients after the IFX therapy compared with patients in the IVIG or VGIH groups. IFX or IFX plus IVIG exhibited improved clinical efficacy in the treatment of KD compared with that of IVIG or VGIH. However, as a limited number of studies was included in the current study, the findings should be verified further.

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Infliximab Treatment for Intravenous Immunoglobulin-resistant Kawasaki Disease: a Multicenter Study in Korea

Cited by 23 publications

References 25 publications

Resolution of Giant Coronary Aneurisms in a Child With Refractory Kawasaki Disease Treated With Anakinra

Resolution of Giant Coronary Aneurisms in a Child With Refractory Kawasaki Disease Treated With Anakinra

Complete Regression of Giant Aneurysms in an Infant with Delayed Diagnosis and Refractory Kawasaki Disease via Combination Anticytokine Therapy: Case Report and Review of Similar Cases

Efficacy of infliximab in the treatment of Kawasaki disease: A systematic review and meta‑analysis

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