ArticleTherapeutic advances in musculoskeletal disease2020
Efficacy of high-dose anakinra in refractory macrophage activation syndrome in adult-onset Still's disease: when dosage matters in overcoming secondary therapy resistance.
Article in Therapeutic advances in musculoskeletal disease, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 11 papers, 1 of them a synthesis that pooled it.
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Who cites it
11 citing papers in PubMed, 1 synthesis or guideline pooled it, 19 citations in OpenAlex.
- Immunopathology of and potential therapeutics for secondary hemophagocytic lymphohistiocytosis/macrophage activation syndrome: a translational perspective.Experimental & molecular medicine · 2024Pooled it
- Rapid response to high-dose Anakinra in refractory SLE-associated Haemophagocytic Lymphohistiocytosis: a case report.Oxford medical case reports · 2026Article
- A Cytokine Storm Unveiled: Diagnostic Challenges of Macrophage Activation Syndrome in the Context of Undiagnosed Systemic Lupus Erythematosus in an Adolescent.Annals of African medicine · 2026Article
- Macrophage activation syndrome-associated adult onset Still disease treatment: a scoping review of case reports and case series.Proceedings (Baylor University. Medical Center) · 2025Article
- The NLRP3 inflammasome is essential for IL-18 production in a murine model of macrophage activation syndrome.Disease models & mechanisms · 2024Article
- The NLRP3 inflammasome is essential for IL-18 production in a murine model of macrophage activation syndrome.bioRxiv : the preprint server for biology · 2024Article
- A Prospective Cohort Study of COVID-19: Evaluation of the Early Role of IL-1 and IL-6 Antagonists in Improving the Outcome of the Illness and Reduction in the Risk of Death.Healthcare (Basel, Switzerland) · 2023Article
- Interferon-γ blockade in CAR T-cell therapy-associated macrophage activation syndrome/hemophagocytic lymphohistiocytosis.Blood advances · 2023Article
- Case Report: Severe Rhabdomyolysis and Multiorgan Failure After ChAdOx1 nCoV-19 Vaccination.Frontiers in immunology · 2022Article
- Anakinra in Paediatric Rheumatology and Periodic Fever Clinics: Is the Higher Dose Safe?Frontiers in pediatrics · 2022Article
- Detection and Prediction of Macrophage Activation Syndrome in Still's Disease.Journal of clinical medicine · 2021Article
Corrections and comments
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Authors and funding
3 authors at 1 institution in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Macrophage activation syndrome (MAS) is a severe, potentially fatal complication of rheumatic diseases. This case demonstrates the significant challenges and therapeutic considerations in adult-onset Still's disease (AOSD) complicated with MAS at initial presentation, which will be discussed. MAS in our patient was refractory to the first-line therapy with high-dose corticosteroids, early administration of anakinra at a standard dosage and subsequent add-on treatments with cyclosporine A, IVIG, etoposides and tocilizumab. At 2 months after presentation, the patient was still critically ill with clinical, laboratory and histological signs of an active uncontrolled MAS. Notably, adoption of anakinra at a high dosage finally induced remission. This case confirms that adjusted dosage of anakinra is an effective therapeutic strategy in a severe AOSD-related MAS. It is tempting to speculate that anakinra at a high dosage, if used earlier, would have significantly changed the course of the disease in our patient and could have led to earlier remission.
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