Usefulness and safety of anakinra in refractory Kawasaki disease complicated by coronary artery aneurysm

Guillaume, Marie-Paule; Reumaux, Héloïse; Dubos, François

doi:10.1017/s1047951117002864

Cited by 48 publications

(42 citation statements)

References 11 publications

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“…Our findings demonstrate that Candida PAMPs initiate arteritis by activating TRMs in the aortic root and inducing chemokine and cytokine production in a mouse model of vasculitis resembling KD. Several case reports have shown promising results using the IL-1R antagonist Anakinra to treat KD (57,58) and new therapeutics that target the CCL2/CCR2 chemokine axis are currently in clinical trials for cancer (59,60). Finally, Syk inhibitors are available and have been in clinical trials for the treatment of rheumatoid arthritis (61).…”

Section: Discussionmentioning

confidence: 99%

Dectin-2–induced CCL2 production in tissue-resident macrophages ignites cardiac arteritis

Miyabe¹,

Miyabe²,

Moreno³

et al. 2019

Journal of Clinical Investigation

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Section: Discussionmentioning

confidence: 99%

Dectin-2–induced CCL2 production in tissue-resident macrophages ignites cardiac arteritis

Miyabe¹,

Miyabe²,

Moreno³

et al. 2019

Journal of Clinical Investigation

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“…Notwithstanding, at least 5% of children remain febrile despite multiple dose of IVIG [18]. These particular patients are at even greater risk of CA complications, and additional therapies are usually administered [19], including corticosteroids [20][21][22], anti-TNF alpha agents [23,24], cyclosporine [25], cyclophosphamide [26] and more recently anakinra (anti-interleukine1) [27,28]. The use of these additional therapies is based on effectiveness in other vasculitis, with no prospective clinical trials to show effectiveness on coronary artery outcome.…”

Section: Discussionmentioning

confidence: 99%

Profile of resistance to IVIG treatment in patients with Kawasaki disease and concomitant infection

Dionne

Poupart

et al. 2018

PLoS ONE

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IntroductionKawasaki disease (KD) can be associated with concomitant viral or bacterial infections. Children with persistent or recurrent fever 36 hours after the end of intravenous immunoglobulin (IVIG) are considered to be resistant to treatment and are at increased risk for coronary complications. Although concomitant infection does not affect coronary outcome, it is unknown how it influences the response to IVIG treatment.MethodologyRetrospective cohort study between 2008 and 2016 in a tertiary pediatric university hospital, including 154 children, of which 59 (38%) had concomitant infection.ResultsChildren with concomitant infection were more likely to have fever 48 hours after initial IVIG treatment (36% vs 20%, p = 0.05) and to be treated with a second dose (33% vs 18%, p = 0.04). Children with infection had higher C-reactive protein at the time of diagnosis (148 vs 112 mg/L, p = 0.04), and 48 hours after IVIG administration (111 vs 59 mg/L, p = 0.003). Nevertheless, there was no statistically significant difference in the prevalence of coronary complications (Z-score > 2.5) between children with and without concomitant infection (36% vs 39%, p = 0.68).ConclusionChildren with KD and concomitant infection are more likely to have persistent fever and elevated inflammatory markers after treatment. This association increases the likelihood of receiving a second dose of IVIG but not the risk of coronary complication. Accordingly, prospective studies to distinguish true IVIG resistance from infection induced persistent fever is warranted.

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“…e final case details response of a child with resistant KD who was then treated with anakinra around one month into disease with significant improvement of z-scores, although not complete normalization. Anakinra treatment in this case was 10 weeks [12]. In contrast to these cases, however a retrospective case series published in January 2018 analyzed multiple cases and found that anakinra showed "neither a striking nor a rapid decrease of coronary dilations" and could not determine if anakinra itself had effects on the CAA [13].…”

Section: Discussionmentioning

confidence: 68%

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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Usefulness and safety of anakinra in refractory Kawasaki disease complicated by coronary artery aneurysm

Cited by 48 publications

References 11 publications

Dectin-2–induced CCL2 production in tissue-resident macrophages ignites cardiac arteritis

Dectin-2–induced CCL2 production in tissue-resident macrophages ignites cardiac arteritis

Profile of resistance to IVIG treatment in patients with Kawasaki disease and concomitant infection

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

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