ArticleRheumatology and therapy2023
Risk of Macrophage Activation Syndrome in Patients with Adult-Onset Still's Disease Treated with IL-1 and IL-6 Inhibitors: A Meta-analysis and Single-Center Experience.
Article in Rheumatology and therapy, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers.
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Who cites it
10 citing papers in PubMed.
- Early C-reactive protein and ferritin kinetics after tocilizumab initiation predict macrophage activation syndrome in Still disease: a single-centre retrospective cohort study.EULAR rheumatology open · 2026Article
- Diagnostic performance of HScore for macrophage activation syndrome complicating Still's disease in adults: a multicentre case-control study.Rheumatology (Oxford, England) · 2026Article
- Macrophage Activation Syndrome Associated with Adult-onset Still's Disease Successfully Treated with Plasma Exchange and Canakinumab.Internal medicine (Tokyo, Japan) · 2026Article
- Beyond Hyperferritinemia: Evaluating Ferritin as a Predictor of Advanced Therapy in Adult-Onset Still's Disease.Diagnostics (Basel, Switzerland) · 2025Article
- A case of adult-onset Still disease complicated by macrophage activation syndrome and toxic epidermal necrolysis.EULAR rheumatology open · 2025Article
- nCD64 index as a new early predictive biomarker for macrophage activation syndrome in adult-onset still's disease.Arthritis research & therapy · 2025Article
- Interleukin-6 Immunology in Macrophage Activation Syndrome Related to Neuroimmunological Diseases.Internal medicine (Tokyo, Japan) · 2025Article
- Adult-Onset Still's Disease Presenting as Aseptic Meningitis: A Case Report and Literature Review.Cureus · 2025Article
- Marked exacerbation of resolving macrophage activation syndrome in a patient with adult-onset Still's disease following one dose of granulocyte colony-stimulating factor.BMJ case reports · 2025Article
- Adult-Onset Still's Disease (AOSD)-On the Basis of Own Cases.Biomedicines · 2024Article
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Abstract
introductionPatients with adult-onset Still's disease (AOSD) are at risk of developing macrophage activation syndrome (MAS), a life-threatening condition. Some cases of MAS have been reported following the use of biological agents, highlighting the need to identify contributing factors. This study aims to examine the characteristics of MAS in patients with AOSD treated with anakinra (ANA) or tocilizumab (TCZ).
methodsA systematic search was conducted across four online databases to identify studies reporting the incidence rates of MAS in patients with AOSD treated with ANA or TCZ. Meta-analysis was performed using a random-effects model and the generic inverse variance method to estimate the pooled incidence rates. The difference in incidence rates of MAS between TCZ and ANA was assessed. Additionally, we analyzed laboratory data and clinical features of AOSD cases at our institution, stratifying them into two groups: those who developed MAS after TCZ administration and those who did not.
resultsOf the 455 screened articles, we included five ANA and six TCZ studies. The pooled incidence rates of MAS were 1.50% (95% confidence interval [CI], 0-3.36) for ANA (345 patients) and 14.01% (95% CI 4.51-23.51) for TCZ (94 patients). MAS incidence was significantly higher in the TCZ group (P = 0.01). Among the 17 patients from our institution, the six patients who developed MAS had significantly higher white blood cell and neutrophil counts, as well as elevated levels of lactate dehydrogenase, C-reactive protein, and ferritin before TCZ induction (P < 0.05).
conclusionsIn patients with AOSD, the manifestation of MAS is influenced by multiple causative factors. Consequently, the administration of TCZ should be approached with caution, particularly in patients exhibiting elevated inflammatory markers.
trial registrationThis study was registered with the Clinical Trial Registry of the University Hospital Medical Information Network (Japan) as UMIN000049243.
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