Successful treatment of macrophage activation syndrome complicating adult Still disease with anakinra
A previously healthy 20‐year‐old man presented with adult Still disease (ASD). He developed life‐threatening macrophage activation syndrome (MAS), which was refractory to standard immunosuppression but responded dramatically to the IL‐1 receptor antagonist anakinra. Subsequent immunological investig...
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| Published in | Internal medicine journal Vol. 42; no. 12; pp. 1358 - 1362 |
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| Main Authors | , , , |
| Format | Journal Article |
| Language | English |
| Published |
Australia
Blackwell Publishing Ltd
01.12.2012
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| Abstract | A previously healthy 20‐year‐old man presented with adult Still disease (ASD). He developed life‐threatening macrophage activation syndrome (MAS), which was refractory to standard immunosuppression but responded dramatically to the IL‐1 receptor antagonist anakinra. Subsequent immunological investigations included assessment of the perforin expression of natural killer (NK) cells and CD8+ T cells, which confirmed MAS. |
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| AbstractList | A previously healthy 20-year-old man presented with adult Still disease (ASD). He developed life-threatening macrophage activation syndrome (MAS), which was refractory to standard immunosuppression but responded dramatically to the IL-1 receptor antagonist anakinra. Subsequent immunological investigations included assessment of the perforin expression of natural killer (NK) cells and CD8+ T cells, which confirmed MAS. |
| Author | Lucas, M. Fernandez, S. Loh, N. K. Prentice, D. |
| Author_xml | – sequence: 1 givenname: N. K. surname: Loh fullname: Loh, N. K. email: nelson.loh@health.wa.gov.au organization: Department of Internal Medicine, Royal Perth Hospital, Western Australia, Perth, Australia – sequence: 2 givenname: M. surname: Lucas fullname: Lucas, M. organization: Department of Immunology and Pathwest Laboratory Medicine, Royal Perth Hospital, Perth, Western Australia, Australia – sequence: 3 givenname: S. surname: Fernandez fullname: Fernandez, S. organization: School of Pathology and Laboratory Medicine, University of Western Australia, Western Australia, Perth, Australia – sequence: 4 givenname: D. surname: Prentice fullname: Prentice, D. organization: Department of Internal Medicine, Royal Perth Hospital, Western Australia, Perth, Australia |
| BackLink | https://www.ncbi.nlm.nih.gov/pubmed/23253002$$D View this record in MEDLINE/PubMed |
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| References_xml | – volume: 65 start-page: 1596 year: 2006 end-page: 1601 article-title: Reactive haemophagocytic syndrome in adult‐onset Still's disease: a report of six patients and a review of the literature publication-title: Ann Rheum Dis – volume: 42 start-page: 375 year: 2003 end-page: 379 article-title: Reduced perforin expression in systemic juvenile idiopathic arthritis is restored by autologous stem cell transplantation publication-title: Rheumatology – volume: 15 start-page: 587 year: 2003 end-page: 590 article-title: Macrophage activation syndrome and reactive hemophagocytic lymphohistiocytosis: the same entities? publication-title: Curr Opin Rheumatol – volume: 34 start-page: 888 year: 1995 article-title: Successful management of reactive haemophagocytic syndrome in systemic onset juvenile chronic arthritis publication-title: Br J Rheumatol – volume: 201 start-page: 1355 year: 2005 end-page: 1359 article-title: Blocking IL‐1 in systemic inflammation publication-title: J Exp Med – volume: 44 start-page: 465 year: 1998 end-page: 468 article-title: Defective natural killer cell function in patients with hemophagocytic lymphohistiocytosis and in first degree relatives publication-title: Pediatr Res – volume: 129 start-page: 750 year: 1996 end-page: 754 article-title: Efficacy of cyclosporine A in the treatment of macrophage activation syndrome in juvenile arthritis: report of five cases publication-title: J Pediatr – volume: 68 start-page: 4 year: 2001 end-page: 10 article-title: Hyperferritinemia as indicator for intravenous immunoglobulin treatment in reactive macrophage activation syndromes publication-title: Am J Hematol – volume: 63 start-page: 281 issue: Suppl year: 2004 article-title: Anakinra in the treatment of adult onset Still's disease publication-title: Ann Rheum Dis – volume: 7 start-page: R30 year: 2005 end-page: 37 article-title: Natural killer cell dysfunction is a distinguishing feature of systemic onset juvenile rheumatoid arthritis and macrophage activation syndrome publication-title: Arthritis Res Ther – volume: 4 start-page: 615 year: 2008 end-page: 620 article-title: A case of macrophage activation syndrome successfully treated with anakinra publication-title: Nat Clin Pract Rheumatol – volume: 51 start-page: 402 year: 2008 end-page: 404 article-title: The role of the initial bone marrow aspirate in the diagnosis of hemophagocytic lymphohistiocytosis publication-title: Pediatr Blood Cancer – volume: 31 start-page: 2071 year: 2004 end-page: 2075 article-title: Effective use of the recombinant interleukin 1 receptor antagonist anakinra in therapy resistant systemic onset juvenile rheumatoid arthritis publication-title: J Rheumatol – volume: 30 start-page: 401 year: 2003 end-page: 403 article-title: Etanercept in treatment of macrophage activation syndrome publication-title: J Rheumatol – volume: 201 start-page: 1279 year: 2005 end-page: 1286 article-title: Role of interleukin‐1 (IL‐1) in the pathogenesis of systemic onset juvenile arthritis and clinical response to IL‐1 blockade publication-title: J Exp Med – volume: 18 start-page: 340 year: 1996 end-page: 345 article-title: Characteristic immune abnormalities in hemophagocytic lymphohistiocytosis publication-title: J Pediatr Hematol Oncol – volume: 126 start-page: 458 year: 2009 end-page: 465 article-title: CD56bright natural killer (NK) cells: an important NK cell subset publication-title: Immunology – volume: 142 start-page: 292 year: 2003 end-page: 296 article-title: Natural killer cell dysfunction in patients with systemic‐onset juvenile rheumatoid arthritis and macrophage activation syndrome publication-title: J Pediatr – volume: 33 start-page: 2081 year: 2006 end-page: 2084 article-title: Interleukin 1 receptor antagonist to treat cytophagic histiocytic panniculitis with secondary hemophagocytic lymphohistiocytosis publication-title: J Rheumatol – volume: 30 start-page: 401 year: 2003 end-page: 403 article-title: Macrophage activation syndrome following initiation of etanercept in a child with systemic onset juvenile rheumatoid arthritis publication-title: J Rheumatol – volume: 28 start-page: 322 year: 2001 end-page: 329 article-title: Diagnostic value of ferritin and glycosylated ferritin in adult onset Still's disease publication-title: J Rheumatol – volume: 64 start-page: 647 year: 2005 end-page: 648 article-title: Refractory adult onset Still's disease successfully treated with anakinra publication-title: Ann Rheum Dis – volume: 32 start-page: 119 year: 2003 end-page: 121 article-title: Bone marrow findings in patients with adult Still's disease publication-title: Scand J Rheumatol – volume: 360 start-page: 2467 year: 2009 end-page: 2470 article-title: Interleukin‐1beta and the autoinflammatory diseases publication-title: N Engl J Med – volume: 52 start-page: 1794 year: 2005 end-page: 1803 article-title: Rapid responses to anakinra in patients with refractory adult‐onset Still's disease publication-title: Arthritis Rheum |
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| Snippet | A previously healthy 20‐year‐old man presented with adult Still disease (ASD). He developed life‐threatening macrophage activation syndrome (MAS), which was... A previously healthy 20-year-old man presented with adult Still disease (ASD). He developed life-threatening macrophage activation syndrome (MAS), which was... |
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| SubjectTerms | adult Still's disease anakinra Antirheumatic Agents - administration & dosage Antirheumatic Agents - therapeutic use Diagnosis, Differential Flow Cytometry haemophagocytic lymphohistiocytosis Humans Interleukin 1 Receptor Antagonist Protein - administration & dosage Interleukin 1 Receptor Antagonist Protein - therapeutic use Killer Cells, Natural Lymphocyte Count macrophage activation syndrome Macrophage Activation Syndrome - complications Macrophage Activation Syndrome - diagnosis Macrophage Activation Syndrome - drug therapy Macrophage Activation Syndrome - immunology Male natural killer cell Still's Disease, Adult-Onset - complications Still's Disease, Adult-Onset - diagnosis Still's Disease, Adult-Onset - immunology Young Adult |
| Title | Successful treatment of macrophage activation syndrome complicating adult Still disease with anakinra |
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