タクロリムスが著効した成人スティル病の１例
症例は70歳，女性で，38度以上の稽留熱が３週間続き，四肢を中心とした紅斑，骨びらんを伴う多関節痛，リウマトイド因子陰性，抗CCP抗体陰性，血清フェリチン値4450ng/mlと上昇し，臨床的兆候や血液検査結果等を勘案し，成人スティル病（adult onset Still’s disease: AOSD）と診断した．プレドニゾロン（PSL）40mg/日で内服治療を開始した．初期治療で速やかに解熱したが，PSL 25mg/日まで減量した時点で発熱，関節痛の再燃がみられ，炎症反応も軽度上昇した．タクロリムス（tacrolimus: TAC）0.5mg/日を併用し，炎症反応は陰性化して軽快した．TAC...
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| Published in | 臨床リウマチ Vol. 27; no. 3; pp. 198 - 204 |
|---|---|
| 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.27.198 |
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| Abstract | 症例は70歳，女性で，38度以上の稽留熱が３週間続き，四肢を中心とした紅斑，骨びらんを伴う多関節痛，リウマトイド因子陰性，抗CCP抗体陰性，血清フェリチン値4450ng/mlと上昇し，臨床的兆候や血液検査結果等を勘案し，成人スティル病（adult onset Still’s disease: AOSD）と診断した．プレドニゾロン（PSL）40mg/日で内服治療を開始した．初期治療で速やかに解熱したが，PSL 25mg/日まで減量した時点で発熱，関節痛の再燃がみられ，炎症反応も軽度上昇した．タクロリムス（tacrolimus: TAC）0.5mg/日を併用し，炎症反応は陰性化して軽快した．TAC併用後はステロイドのさらなる漸減が可能となり，再燃なく最終的にステロイドを中止できた．TACはAOSDでの治療選択肢のひとつで，ステロイド減量困難例でTACが有効な症例があると予想され，今後の症例の積み重ねと解析が必要である． |
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| AbstractList | A 70-year-old woman had high fever for 21 days accompanied by erythema concentrated on extremity, polyarthralgia with bone erosion and serum ferritin was 4450 ng/ml, whereas rheumatoid factor and anti-cyclic citrullinated peptide antibody were negative. We diagnosed as AOSD, initially she was treated with high dose corticosteroids (1 mg/kg/day, prednisone 40 mg) and her symptom were improve. We thought AOSD flare with decreasing the dose of prednisone to 25 mg, when her symptom got worse as pyrexia, arthralgia, and elevation of inflammatory reaction. She was treated to add on tacrolimus (TAC), then it got improved for one week. Finally, we discontinued steroid without relapse AOSD, adding TAC made it possible for steroid tapering. We need to think that TAC is regarded as one of the choice of treatment for AOSD in case of difficulty toward steroid sparing.
症例は70歳，女性で，38度以上の稽留熱が３週間続き，四肢を中心とした紅斑，骨びらんを伴う多関節痛，リウマトイド因子陰性，抗CCP抗体陰性，血清フェリチン値4450ng/mlと上昇し，臨床的兆候や血液検査結果等を勘案し，成人スティル病（adult onset Still’s disease: AOSD）と診断した．プレドニゾロン（PSL）40mg/日で内服治療を開始した．初期治療で速やかに解熱したが，PSL 25mg/日まで減量した時点で発熱，関節痛の再燃がみられ，炎症反応も軽度上昇した．タクロリムス（tacrolimus: TAC）0.5mg/日を併用し，炎症反応は陰性化して軽快した．TAC併用後はステロイドのさらなる漸減が可能となり，再燃なく最終的にステロイドを中止できた．TACはAOSDでの治療選択肢のひとつで，ステロイド減量困難例でTACが有効な症例があると予想され，今後の症例の積み重ねと解析が必要である． 症例は70歳，女性で，38度以上の稽留熱が３週間続き，四肢を中心とした紅斑，骨びらんを伴う多関節痛，リウマトイド因子陰性，抗CCP抗体陰性，血清フェリチン値4450ng/mlと上昇し，臨床的兆候や血液検査結果等を勘案し，成人スティル病（adult onset Still’s disease: AOSD）と診断した．プレドニゾロン（PSL）40mg/日で内服治療を開始した．初期治療で速やかに解熱したが，PSL 25mg/日まで減量した時点で発熱，関節痛の再燃がみられ，炎症反応も軽度上昇した．タクロリムス（tacrolimus: TAC）0.5mg/日を併用し，炎症反応は陰性化して軽快した．TAC併用後はステロイドのさらなる漸減が可能となり，再燃なく最終的にステロイドを中止できた．TACはAOSDでの治療選択肢のひとつで，ステロイド減量困難例でTACが有効な症例があると予想され，今後の症例の積み重ねと解析が必要である． |
| Author | 中村, 正 下村, 泰三 鈴島, 仁 藤本, 哲広 高岡, 宏和 |
| Author_FL | Shimomura Taizo Fujimoto Tetsuhiro Takaoka Hirokazu Nakamura Tadashi Suzushima Hitoshi |
| Author_FL_xml | – sequence: 1 fullname: Takaoka Hirokazu – sequence: 2 fullname: Fujimoto Tetsuhiro – sequence: 3 fullname: Shimomura Taizo – sequence: 4 fullname: Suzushima Hitoshi – sequence: 5 fullname: Nakamura Tadashi |
| Author_xml | – sequence: 1 fullname: 下村, 泰三 organization: くまもと森都総合病院血液内科 – sequence: 1 fullname: 高岡, 宏和 organization: くまもと森都総合病院リウマチ膠原病内科 – sequence: 1 fullname: 中村, 正 organization: くまもと森都総合病院リウマチ膠原病内科 – sequence: 1 fullname: 鈴島, 仁 organization: くまもと森都総合病院血液内科 – sequence: 1 fullname: 藤本, 哲広 organization: くまもと森都総合病院血液内科 |
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| References | 6) Perez C, Artala V: Adult Still’s disease associated with Mycoplasma pneumoniae infection. Clin Infect Dis, 32:E105-106, 2001. 8) Rodeghero R, Cao Y, Olalekan SA, et al: Location of CD4+ T cell priming regulates the differentiation of Th1 and Th17 cells and their contribution to arthritis. J Immunol, 11:5423-5435, 2013. 14) Murakami K, Fujii T, Yukawa N, et al: Successful treatment of a patient with refractory adult Still’s disease by tacrolimus. Mod Rheumatol, 17:167-170, 2007. 5) van de Putte LB, Wouters JM: Adult-onset Still’s disease. Baillieres Clin Rheumatol, 5:263-275, 1991. 9) Sakai R, Nagasawa H, Nishi E, et al: Successful treatment of adult-onset Still’s disease with tocilizumab monotherapy: two case reports and review. Clin Rheumatol, 31:569-574, 2012. 2) Ohta A, Yamaguchi M, Kaneoka H, et al: Adult Still’s disease: review of 228 cases from the literature. J Rheumatol, 14:1139-1146, 1987. 11) Okamura H, Tsutsi H, Komatsu T, et al: Cloning of a new cytokine that induces IFN-γ production by T cells. Nature, 378:88-91, 1995. 1) Efthimiou P, Paik PK, Bielory L: Diagnosis and management of adult onset still’s disease. Ann Rheum Dis, 65:564-572, 2006. 4) Yamaguchi M, Ohta A, Tsunematsu T, et al: Preliminary criteria for classification of adult Still’s disease. J Rheumatol, 19:424-430, 1992. 12) Kuninose M, Iwagaki H, Morimoto Y, et al: Calcineurin antagonists inhibit interferon-gamma production by downregulation of interleukin-18 in human mixed lymphocyte reactions. Acta Medica Okayama, 54:201-209, 2000. 13) Nakamura H, Odani T, Hisada R, et al: Usefulness of tacrolimus for refractory Adult-Onset Still Disease. Mod Rheumatol, Supple S65, 2013. 10) 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. 7) Hoshino T, Ohta A, Yang D, et al: Elevated serum interleukin 6, interferon-gamma, and tumor necrosis factor-alpha levels in patients with adult Still’s disease. J Rheumatol, 25:396-398, 1998. 3) Yamamoto T: Cutaneous manifestations associated with adult-onset Still’s disease: important diagnostic values. Rheumatol Int, 32:2233-2237, 2012. |
| References_xml | – reference: 11) Okamura H, Tsutsi H, Komatsu T, et al: Cloning of a new cytokine that induces IFN-γ production by T cells. Nature, 378:88-91, 1995. – reference: 14) Murakami K, Fujii T, Yukawa N, et al: Successful treatment of a patient with refractory adult Still’s disease by tacrolimus. Mod Rheumatol, 17:167-170, 2007. – reference: 9) Sakai R, Nagasawa H, Nishi E, et al: Successful treatment of adult-onset Still’s disease with tocilizumab monotherapy: two case reports and review. Clin Rheumatol, 31:569-574, 2012. – reference: 10) 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: 1) Efthimiou P, Paik PK, Bielory L: Diagnosis and management of adult onset still’s disease. Ann Rheum Dis, 65:564-572, 2006. – reference: 4) Yamaguchi M, Ohta A, Tsunematsu T, et al: Preliminary criteria for classification of adult Still’s disease. J Rheumatol, 19:424-430, 1992. – reference: 5) van de Putte LB, Wouters JM: Adult-onset Still’s disease. Baillieres Clin Rheumatol, 5:263-275, 1991. – reference: 2) Ohta A, Yamaguchi M, Kaneoka H, et al: Adult Still’s disease: review of 228 cases from the literature. J Rheumatol, 14:1139-1146, 1987. – reference: 13) Nakamura H, Odani T, Hisada R, et al: Usefulness of tacrolimus for refractory Adult-Onset Still Disease. Mod Rheumatol, Supple S65, 2013. – reference: 6) Perez C, Artala V: Adult Still’s disease associated with Mycoplasma pneumoniae infection. Clin Infect Dis, 32:E105-106, 2001. – reference: 8) Rodeghero R, Cao Y, Olalekan SA, et al: Location of CD4+ T cell priming regulates the differentiation of Th1 and Th17 cells and their contribution to arthritis. J Immunol, 11:5423-5435, 2013. – reference: 7) Hoshino T, Ohta A, Yang D, et al: Elevated serum interleukin 6, interferon-gamma, and tumor necrosis factor-alpha levels in patients with adult Still’s disease. J Rheumatol, 25:396-398, 1998. – reference: 3) Yamamoto T: Cutaneous manifestations associated with adult-onset Still’s disease: important diagnostic values. Rheumatol Int, 32:2233-2237, 2012. – reference: 12) Kuninose M, Iwagaki H, Morimoto Y, et al: Calcineurin antagonists inhibit interferon-gamma production by downregulation of interleukin-18 in human mixed lymphocyte reactions. Acta Medica Okayama, 54:201-209, 2000. |
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| Snippet | 症例は70歳，女性で，38度以上の稽留熱が３週間続き，四肢を中心とした紅斑，骨びらんを伴う多関節痛，リウマトイド因子陰性，抗CCP抗体陰性，血清フェリチン... A 70-year-old woman had high fever for 21 days accompanied by erythema concentrated on extremity, polyarthralgia with bone erosion and serum ferritin was... |
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| Title | タクロリムスが著効した成人スティル病の１例 |
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