トシリズマブによりステロイドから離脱し得た重症成人発症スティル病の一例
43歳，女性．発熱，顔面と体幹部の紅斑，関節痛を示す成人発症スティル病に対して，プレドニゾロンの投与が行われたが，治療抵抗性であり，播種性血管内凝固を併発した．血漿交換を施行，シクロフォスファミド，シクロスポリンを追加し，病状は改善した．その後，寛解維持目的としてトシリズマブを導入し，疾患活動性の低下を認めた. プレドニゾロン，シクロスポリンを漸減, 中止したが寛解状態は維持できた．トシリズマブの寛解維持における意義について検討した．...
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| Published in | 臨床リウマチ Vol. 27; no. 3; pp. 205 - 211 |
|---|---|
| Main Authors | , , , , , , , , , , , , |
| Format | Journal Article |
| Language | Japanese |
| Published |
一般社団法人 日本臨床リウマチ学会
2015
The Japanese Society for Clinical Rheumatology and Related Research |
| Subjects | |
| Online Access | Get full text |
| ISSN | 0914-8760 2189-0595 |
| DOI | 10.14961/cra.27.205 |
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| Abstract | 43歳，女性．発熱，顔面と体幹部の紅斑，関節痛を示す成人発症スティル病に対して，プレドニゾロンの投与が行われたが，治療抵抗性であり，播種性血管内凝固を併発した．血漿交換を施行，シクロフォスファミド，シクロスポリンを追加し，病状は改善した．その後，寛解維持目的としてトシリズマブを導入し，疾患活動性の低下を認めた. プレドニゾロン，シクロスポリンを漸減, 中止したが寛解状態は維持できた．トシリズマブの寛解維持における意義について検討した． |
|---|---|
| AbstractList | In recent years, it has been reported that tocilizumab is effective in adult onset Still’s disease (AOSD). We report a case of severe AOSD where tocilizumab was effective and contributed to cessation of steroid treatment. A 43 year-old female patient suffering from fever, skin rash and arthralgia was diagnosed as having AOSD. Despite treatment with high-dose prednisolone, its response was poor and the patient suffered the complications of disseminated intravascular coagulation (DIC) and acute kidney injury (AKI). After disease activity improved by plasma exchange, cyclosporine and cyclophosphamide, tocilizumab was introduced with the aim of maintaining remission. Thereafter, disease activity stayed low even after cessation of prednisolone and cyclosporine. The significance of tocilizumab as the maintenance therapy is discussed along with previous reports.
43歳，女性．発熱，顔面と体幹部の紅斑，関節痛を示す成人発症スティル病に対して，プレドニゾロンの投与が行われたが，治療抵抗性であり，播種性血管内凝固を併発した．血漿交換を施行，シクロフォスファミド，シクロスポリンを追加し，病状は改善した．その後，寛解維持目的としてトシリズマブを導入し，疾患活動性の低下を認めた. プレドニゾロン，シクロスポリンを漸減, 中止したが寛解状態は維持できた．トシリズマブの寛解維持における意義について検討した． 43歳，女性．発熱，顔面と体幹部の紅斑，関節痛を示す成人発症スティル病に対して，プレドニゾロンの投与が行われたが，治療抵抗性であり，播種性血管内凝固を併発した．血漿交換を施行，シクロフォスファミド，シクロスポリンを追加し，病状は改善した．その後，寛解維持目的としてトシリズマブを導入し，疾患活動性の低下を認めた. プレドニゾロン，シクロスポリンを漸減, 中止したが寛解状態は維持できた．トシリズマブの寛解維持における意義について検討した． |
| Author | 李, 進海 樋野, 尚一 朝戸, 佳世 野﨑, 祐史 船内, 正憲 井上, 明日圭 岸本, 和也 矢野, 智洋 伊丹, 哲 松村, 到 志賀, 俊彦 田崎, 知江美 酒井, 健史 |
| Author_FL | Itami Tetu Ri Shinkai Kishimoto Kazuya Matsumura Itaru Shiga Toshihiko Sakai Kenji Hino Shoichi Yano Tomohiro Asato Kayo Tasaki Chiemi Funauchi Masanori Nozaki Yuji inoue Asuka |
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| DocumentTitleAlternate | A case of severe adult onset Still’s disease that became free from steroid therapy by introduction tocilizumab |
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| References | 1) Ohta A, Yamaguchi M, Tsunem T, et al: Adult Still’s disease: a multicenter survey of Japanese pations. J Rheumatol, 17:1058-1063, 1990. 13) Ortiz-Sanjuan F, Blanco R, Calvo-Rio Y, et al: Efficacy of Tocilizumab in Conventional Treatment-Refractory Adult-Onset Still’s Disease Multicenter Retrospective Open-Label Study of Thirty-Four Patients. Arthritis Rheum, 66:1659-1665, 2014. 3) Marchesoni A, Ceravolo GP, Battafarano N, et al: Cyclosporin A in the treatment of adult onset still’s disease. J Rheumatol, 24: 1582-1587, 1997. 4) Hubert de Boysson, Jerome Fevrier, Amelie Nicolle, et al: Tocilizumab in the treatment of the adult-onset Still’s disease: current clinical evidence. Clin Rheumatol, 32:141-147, 2013. 10) 太田明英：膠原病診療の実際 成人Still病．臨牀と研究, 87：1219-1224, 2010. 8) Efthimiou P, Kontzias A, Ward CM, et al: Adult-onset Still’s disease:can recent advances in our understanding of its pathogenesis lead to targeted therapy? Nat Clin Pract Rheumatol, 3:328-335, 2007. 11) Fautrel B:Adult-onset Still disease. Best Pract Res Clin Rheumatol, 22:773-792, 2008. 9) Kawashima M, Yamamura M, Taniai M, et al: Levels of interleukin-18 and its binding inhibitors in the blood circulation of patients with adult-onset Still’s disease. Arthritis Rheum, 44:550-560, 2001. 15) Shimizu M, Nakagishi Y, Kasai K, et al: Tocilizumab masks the clinical symptoms of systemic juvenile idiopathic arthritis-associated macrophage activation syndrome: the diagnostic significance of interleukin-18 and interleukin-6. Cytokine, 58: 287-294, 2012. 6) Kraetsch HG, Antoni C, Kalden JR, et al: Successful treatment of a small cohort of patients with adult onset of Still’s disease with infliximab: first experiences. Ann Rheum Dis, 60 Suppl 3:iii55-iii57, 2001. 14) Nishina N, Kaneko Y, Kameda H, et al: The effect of tocilizumab on preventing relapses in adult-onset Still’s disease: A retrospective, single-center study. Mod Rheumatol, 17:1-4, 2014. 16) Masafumi K, Yuko T, Hiroyuki Y, et al: Benefit and a possible risk of tocilizumab therapy for adult-onset Still disease accompanied by macrophage-activation syndrome. Mod Rheumatol, 21:92-96, 2011. 7) Yamaguchi M, Ohta A, Tsunem T, et al: Preliminary criteria for classification of adult Still’s disease. J Rheumatol, 19:424-430, 1992. 12) Benedetti F, Brunner HI, Ruperto N, et al: Randomized Trial of Tocilizumab in Systemic Juvenile Idiopathic Arthritis. N Engl J Med, 367; 25:2385-2395, 2012. 2) Fautrel B, Borget C, Rozenberg S, et al: Corticosteroid sparing effect of low dose methotrexate treatment in adult Still’s diase. J Rheumatol, 26:373-378, 1999. 5) Puechal X, DeBandt M, Berthelot JM, et al: Tocilizumab in refractory adult Still’s disease. Arthritis Care Res, 63:155-159, 2011. 17) 高橋裕樹，鈴木知佐子，山本元久：成人Still病の治療の実際．Medical Practice, 28:1269-1274, 2011. |
| References_xml | – reference: 15) Shimizu M, Nakagishi Y, Kasai K, et al: Tocilizumab masks the clinical symptoms of systemic juvenile idiopathic arthritis-associated macrophage activation syndrome: the diagnostic significance of interleukin-18 and interleukin-6. Cytokine, 58: 287-294, 2012. – reference: 6) Kraetsch HG, Antoni C, Kalden JR, et al: Successful treatment of a small cohort of patients with adult onset of Still’s disease with infliximab: first experiences. Ann Rheum Dis, 60 Suppl 3:iii55-iii57, 2001. – reference: 5) Puechal X, DeBandt M, Berthelot JM, et al: Tocilizumab in refractory adult Still’s disease. Arthritis Care Res, 63:155-159, 2011. – reference: 2) Fautrel B, Borget C, Rozenberg S, et al: Corticosteroid sparing effect of low dose methotrexate treatment in adult Still’s diase. J Rheumatol, 26:373-378, 1999. – reference: 7) Yamaguchi M, Ohta A, Tsunem T, et al: Preliminary criteria for classification of adult Still’s disease. J Rheumatol, 19:424-430, 1992. – reference: 3) Marchesoni A, Ceravolo GP, Battafarano N, et al: Cyclosporin A in the treatment of adult onset still’s disease. J Rheumatol, 24: 1582-1587, 1997. – reference: 10) 太田明英：膠原病診療の実際 成人Still病．臨牀と研究, 87：1219-1224, 2010. – reference: 9) Kawashima M, Yamamura M, Taniai M, et al: Levels of interleukin-18 and its binding inhibitors in the blood circulation of patients with adult-onset Still’s disease. Arthritis Rheum, 44:550-560, 2001. – reference: 12) Benedetti F, Brunner HI, Ruperto N, et al: Randomized Trial of Tocilizumab in Systemic Juvenile Idiopathic Arthritis. N Engl J Med, 367; 25:2385-2395, 2012. – reference: 14) Nishina N, Kaneko Y, Kameda H, et al: The effect of tocilizumab on preventing relapses in adult-onset Still’s disease: A retrospective, single-center study. Mod Rheumatol, 17:1-4, 2014. – reference: 17) 高橋裕樹，鈴木知佐子，山本元久：成人Still病の治療の実際．Medical Practice, 28:1269-1274, 2011. – reference: 4) Hubert de Boysson, Jerome Fevrier, Amelie Nicolle, et al: Tocilizumab in the treatment of the adult-onset Still’s disease: current clinical evidence. Clin Rheumatol, 32:141-147, 2013. – reference: 13) Ortiz-Sanjuan F, Blanco R, Calvo-Rio Y, et al: Efficacy of Tocilizumab in Conventional Treatment-Refractory Adult-Onset Still’s Disease Multicenter Retrospective Open-Label Study of Thirty-Four Patients. Arthritis Rheum, 66:1659-1665, 2014. – reference: 1) Ohta A, Yamaguchi M, Tsunem T, et al: Adult Still’s disease: a multicenter survey of Japanese pations. J Rheumatol, 17:1058-1063, 1990. – reference: 8) Efthimiou P, Kontzias A, Ward CM, et al: Adult-onset Still’s disease:can recent advances in our understanding of its pathogenesis lead to targeted therapy? Nat Clin Pract Rheumatol, 3:328-335, 2007. – reference: 16) Masafumi K, Yuko T, Hiroyuki Y, et al: Benefit and a possible risk of tocilizumab therapy for adult-onset Still disease accompanied by macrophage-activation syndrome. Mod Rheumatol, 21:92-96, 2011. – reference: 11) Fautrel B:Adult-onset Still disease. Best Pract Res Clin Rheumatol, 22:773-792, 2008. |
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| Snippet | 43歳，女性．発熱，顔面と体幹部の紅斑，関節痛を示す成人発症スティル病に対して，プレドニゾロンの投与が行われたが，治療抵抗性であり，播種性血管内凝固を併発した．血漿交換を施行，シクロフォスファミド，シクロスポリンを追加し，病状は改善した．その後，寛解維持目的としてトシリズマブを導入し，疾患活動性の低下を認めた.... In recent years, it has been reported that tocilizumab is effective in adult onset Still’s disease (AOSD). We report a case of severe AOSD where... |
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| SubjectTerms | adult onset Still’s disease macrophage activation syndrome tocilizumab |
| Title | トシリズマブによりステロイドから離脱し得た重症成人発症スティル病の一例 |
| URI | https://www.jstage.jst.go.jp/article/cra/27/3/27_205/_article/-char/ja https://cir.nii.ac.jp/crid/1390001204342668800 |
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