ArticleBMJ case reports2019
High-dose anakinra as treatment for macrophage activation syndrome caused by refractory Kawasaki disease in an infant.
Article in BMJ case reports, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 28 papers, 6 of them syntheses that pooled it.
What it found
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
28 citing papers in PubMed, 6 syntheses or guidelines pooled it, 37 citations in OpenAlex.
- Anakinra for intravenous immunoglobulin-resistant Kawasaki disease: a systematic literature review.RMD open · 2026Pooled it
- Anakinra use in pediatric kawasaki disease: inflammation control, coronary artery outcomes, and safety.Pediatric rheumatology online journal · 2026Pooled it
- Immunopathology of and potential therapeutics for secondary hemophagocytic lymphohistiocytosis/macrophage activation syndrome: a translational perspective.Experimental & molecular medicine · 2024Pooled it
- American College of Rheumatology Clinical Guidance for Multisystem Inflammatory Syndrome in Children Associated With SARS-CoV-2 and Hyperinflammation in Pediatric COVID-19: Version 2.Arthritis & rheumatology (Hoboken, N.J.) · 2021Guideline
- American College of Rheumatology Clinical Guidance for Multisystem Inflammatory Syndrome in Children Associated With SARS-CoV-2 and Hyperinflammation in Pediatric COVID-19: Version 1.Arthritis & rheumatology (Hoboken, N.J.) · 2020Guideline
- A Comprehensive Update on Kawasaki Disease Vasculitis and Myocarditis.Current rheumatology reports · 2020Pooled it
- Retrospective study: clinical analysis of Kawasaki disease complicated with macrophage activation syndrome.Italian journal of pediatrics · 2026Article
- 2016 sJIA-MAS criteria and a step-up therapeutic approach in KD-MAS: insights from a combined cohort and literature review.RMD open · 2026Review
- Emergency in pediatric rheumatology: a narrative review.Frontiers in medicine · 2026Review
- Anakinra for infants under six months with Kawasaki disease and coronary artery lesions: a multicenter case series and literature review.Pediatric rheumatology online journal · 2025Article
- Off-Label Use of Anakinra in Inflammatory Conditions in Neonates and Infants Up to 3 Months of Age: A Case Series and a Review of the Literature.Paediatric drugs · 2025Review
- Macrophage Activation Syndrome in Kawasaki Disease: Insights from a Systematic Literature Review on Diagnosis, Clinical Features, and Treatment.Children (Basel, Switzerland) · 2025Review
- Kawasaki Disease-Associated Cytokine Storm Syndrome.Advances in experimental medicine and biology · 2024Review
- [Clinical features of Kawasaki disease complicated by macrophage activation syndrome: an analysis of 27 cases].Zhongguo dang dai er ke za zhi = Chinese journal of contemporary pediatrics · 2023Article
- Review
- American College of Rheumatology Clinical Guidance for Multisystem Inflammatory Syndrome in Children Associated With SARS-CoV-2 and Hyperinflammation in Pediatric COVID-19: Version 3.Arthritis & rheumatology (Hoboken, N.J.) · 2022Article
- Observational
- NLRP3 inflammasome activation and SARS-CoV-2-mediated hyperinflammation, cytokine storm and neurological syndromes.International journal of physiology, pathophysiology and pharmacology · 2022Review
- Management of COVID-19-associated multisystem inflammatory syndrome in children: A comprehensive literature review.Progress in pediatric cardiology · 2021Review
- COVID-19 associated Multisystem Inflammatory Syndrome in Children (MIS-C) guidelines; revisiting the Western New York approach as the pandemic evolves.Progress in pediatric cardiology · 2021Article
Corrections and comments
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Authors and funding
3 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
We report a 12-week-old boy presenting with incomplete refractory Kawasaki disease (KD) complicated with macrophage activation syndrome (MAS). The infant presented with cerebral irritability, pain, tachypnoea and vomiting for 10 days. He did not fulfil any of the classic diagnostic criteria for KD. Pericardial effusion on echocardiography in addition to severe dilatation of the coronary arteries in combination with leucocytosis and raised acute phase reactants led to the diagnosis of incomplete KD. Treatment with intravenous immunoglobulin and aspirin was initiated but without any response. The condition was subsequently refractory to additional treatment with infliximab and high-dose methylprednisolone. His condition worsened, fulfilling the criteria for MAS. High-dose anakinra was initiated, and remission of the inflammation was achieved.
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Registered trials
Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.