ArticleMedicine2017
Refractory adult-onset Still disease complicated by macrophage activation syndrome and acute myocarditis: A case report treated with high doses (8 mg/kg/d) of anakinra.
Article in Medicine, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 26 papers, 1 of them a synthesis that pooled it.
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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
26 citing papers in PubMed, 1 synthesis or guideline pooled it, 52 citations in OpenAlex.
- Management of adult-onset Still's disease with interleukin-1 inhibitors: evidence- and consensus-based statements by a panel of Italian experts.Arthritis research & therapy · 2019Pooled it
- Efficacy and Safety of Anakinra Plus Standard of Care for Patients With Severe COVID-19: A Randomized Phase 2/3 Clinical Trial.JAMA network open · 2023Trial
- Cardioimmunology of Myocarditis: Targeting the IL-1 Pathway.Current cardiology reports · 2026Review
- Macrophage activation syndrome-associated adult onset Still disease treatment: a scoping review of case reports and case series.Proceedings (Baylor University. Medical Center) · 2025Article
- Treatment of pediatric severe acute myopericarditis with anakinra: a case report and literature review.Frontiers in pediatrics · 2025Article
- Innate and adaptive immunity in acute myocarditis.International journal of cardiology · 2024Review
- IL-1 Family Blockade in Cytokine Storm Syndromes.Advances in experimental medicine and biology · 2024Review
- Cytokine Storm Syndrome Associated with Systemic Juvenile Idiopathic Arthritis.Advances in experimental medicine and biology · 2024Review
- Macrophage Activation Syndrome in the Setting of Rheumatic Diseases.Advances in experimental medicine and biology · 2024Review
- Fulminant myocarditis with adult-onset Still's disease: case-based review.Clinical rheumatology · 2023Review
- Case report: High-risk acute promyelocytic leukemia and COVID-19-related myocarditis one patient, two cytokine storms.Frontiers in oncology · 2023Article
- Secondary hemophagocytic lymphohistiocytosis due to Heartland virus.BMJ case reports · 2022Article
- Effect of COVID-19 on anakinra-induced remission in homozygous STX11 hemophagocytosis lymphohistiocytosis.Pediatric blood & cancer · 2021Article
- An Update on the Pathogenic Role of Macrophages in Adult-Onset Still's Disease and Its Implication in Clinical Manifestations and Novel Therapeutics.Journal of immunology research · 2021Review
- High dose subcutaneous Anakinra to treat acute respiratory distress syndrome secondary to cytokine storm syndrome among severely ill COVID-19 patients.Journal of autoimmunity · 2020Article
- Macrophage activation syndrome associated with adult-onset Still's disease: a multicenter retrospective analysis.Clinical rheumatology · 2020Article
- COVID-19 gone bad: A new character in the spectrum of the hyperferritinemic syndrome?Autoimmunity reviews · 2020Review
- Myocarditis in Adult-Onset Still's Disease: Case-Based Review.Clinical rheumatology · 2020Review
- Efficacy of high-dose anakinra in refractory macrophage activation syndrome in adult-onset Still's disease: when dosage matters in overcoming secondary therapy resistance.Therapeutic advances in musculoskeletal disease · 2020Article
- Case Report: Life-Threatening Macrophage Activation Syndrome With Fulminant Myocarditis Successfully Rescued by High Dose Intravenous Anakinra.Frontiers in pediatrics · 2020Article
Corrections and comments
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Authors and funding
5 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
rationaleMyocarditis is a rare but potentially fatal complication of Still's disease (about 7% of total cases). PATIENT CONCERNS: A 42-year-old woman was admitted to our ward with high-grade fever, rash and polyarthralgia, lasting since 4 weeks and rapidly complicated by MAS and acute heart failure. DIAGNOSES: Adult Onset Still's Disease rapidly developping macrophage activation syndrome and disseminated intravascular coagulopathy, further complicated by iperacute myocarditis with cardiac arrest.
interventionsAfter failure of conventional therapies (steroids plus cyclosporine and then biological therapy with Anakinra 100 mg/day), the patient was treated with anakinra 100 mg sc 1 fl 4 times a day. OUTCOMES: Fast clinical and laboratoristic improvement and subsequent disease remission with complete recovery of cardiac function. LESSONS: This is the first case report in which high doses of Anakinra have been used to treat a refractory AOSD complicated by MAS and myocarditis. In AOSD complicated by life-threatening conditions, probably we need to consider aggressive therapeutic approaches with higher doses of Il-1 receptor blocker to switch off the hyper-inflammation.
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