{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,3,6]],"date-time":"2026-03-06T17:40:42Z","timestamp":1772818842827,"version":"3.50.1"},"reference-count":22,"publisher":"Oxford University Press (OUP)","issue":"2","license":[{"start":{"date-parts":[[2025,1,1]],"date-time":"2025-01-01T00:00:00Z","timestamp":1735689600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,7,25]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n               <jats:p>Tocilizumab (TCZ) is effective for inducing remission in adult-onset Still\u2019s disease (AOSD), but its use may occasionally trigger macrophage activation syndrome (MAS). The rationale for re-introducing TCZ in patients with a history of MAS is not well established. Here, we report a case of successful re-administration of TCZ for an AOSD relapse in a patient with a prior history of MAS during TCZ therapy. A 67-year-old woman, initially treated with TCZ for polyarthritis, developed MAS associated with AOSD. MAS was resolved with glucocorticoid pulse therapy, high-dose glucocorticoids, and cyclosporine A. However, AOSD relapsed during glucocorticoid tapering. Methotrexate, cyclosporine A, and repeated glucocorticoid pulses failed to control the disease. Following another glucocorticoid pulse, TCZ (8\u2009mg\/kg weekly) was re-introduced intravenously. This approach allowed successful glucocorticoid tapering and long-term remission. This case highlights the complexities of managing AOSD: while the initial TCZ therapy may have contributed to the onset of MAS, the subsequent re-introduction of TCZ enabled effective disease control and sustained remission.<\/jats:p>","DOI":"10.1093\/mrcr\/rxaf020","type":"journal-article","created":{"date-parts":[[2025,4,8]],"date-time":"2025-04-08T08:32:35Z","timestamp":1744101155000},"source":"Crossref","is-referenced-by-count":2,"title":["Successful re-administration of tocilizumab in a patient with adult-onset Still\u2019s disease after improvement of macrophage activation syndrome"],"prefix":"10.1093","volume":"9","author":[{"given":"Yuma","family":"Nagasawa","sequence":"first","affiliation":[{"name":"Department of Stem Cell and Immune Regulation, Yokohama City University Graduate School of Medicine , 3-9 Fukuura, Kanazawa-Ku, Yokohama 236-0004,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4965-9274","authenticated-orcid":false,"given":"Kaoru","family":"Takase-Minegishi","sequence":"additional","affiliation":[{"name":"Department of Stem Cell and Immune Regulation, Yokohama City University Graduate School of Medicine , 3-9 Fukuura, Kanazawa-Ku, Yokohama 236-0004,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Soichiro","family":"Adachi","sequence":"additional","affiliation":[{"name":"Department of Stem Cell and Immune Regulation, Yokohama City University Graduate School of Medicine , 3-9 Fukuura, Kanazawa-Ku, Yokohama 236-0004,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Kento","family":"Ichikawa","sequence":"additional","affiliation":[{"name":"Department of Stem Cell and Immune Regulation, Yokohama City University Graduate School of Medicine , 3-9 Fukuura, Kanazawa-Ku, Yokohama 236-0004,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hideto","family":"Nagai","sequence":"additional","affiliation":[{"name":"Department of Stem Cell and Immune Regulation, Yokohama City University Graduate School of Medicine , 3-9 Fukuura, Kanazawa-Ku, Yokohama 236-0004,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Tomoya","family":"Watanabe","sequence":"additional","affiliation":[{"name":"Department of Environmental Immuno-Dermatology, Yokohama City University Graduate School of Medicine , 3-9 Fukuura, Kanazawa-Ku, Yokohama 236-0004,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0297-6627","authenticated-orcid":false,"given":"Yukie","family":"Yamaguchi","sequence":"additional","affiliation":[{"name":"Department of Environmental Immuno-Dermatology, Yokohama City University Graduate School of Medicine , 3-9 Fukuura, Kanazawa-Ku, Yokohama 236-0004,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ryusuke","family":"Yoshimi","sequence":"additional","affiliation":[{"name":"Department of Stem Cell and Immune Regulation, Yokohama City University Graduate School of Medicine , 3-9 Fukuura, Kanazawa-Ku, Yokohama 236-0004,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9488-661X","authenticated-orcid":false,"given":"Yohei","family":"Kirino","sequence":"additional","affiliation":[{"name":"Department of Stem Cell and Immune Regulation, Yokohama City University Graduate School of Medicine , 3-9 Fukuura, Kanazawa-Ku, Yokohama 236-0004,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8967-4954","authenticated-orcid":false,"given":"Hideaki","family":"Nakajima","sequence":"additional","affiliation":[{"name":"Department of Stem Cell and Immune Regulation, Yokohama City University Graduate School of Medicine , 3-9 Fukuura, Kanazawa-Ku, Yokohama 236-0004,","place":["Japan"]}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"286","published-online":{"date-parts":[[2025,4,10]]},"reference":[{"key":"2025080201024117200_R1","doi-asserted-by":"crossref","first-page":"736","DOI":"10.1080\/14397595.2018.1465633","article-title":"Evidence-based clinical practice guideline for adult Still\u2019s disease","volume":"28","author":"Mimura","year":"2018","journal-title":"Mod Rheumatol"},{"key":"2025080201024117200_R2","doi-asserted-by":"crossref","first-page":"603","DOI":"10.1038\/s41584-018-0081-x","article-title":"Mechanisms, biomarkers and targets for adult-onset Still\u2019s disease","volume":"14","author":"Feist","year":"2018","journal-title":"Nat Rev Rheumatol"},{"key":"2025080201024117200_R3","doi-asserted-by":"crossref","first-page":"1614","DOI":"10.1136\/ard-2024-225851","article-title":"EULAR\/PReS recommendations for the diagnosis and management of Still\u2019s disease, comprising systemic juvenile idiopathic arthritis and adult-onset Still\u2019s disease","volume":"83","author":"Fautrel","year":"2024","journal-title":"Ann Rheum Dis"},{"key":"2025080201024117200_R4","doi-asserted-by":"crossref","first-page":"1271","DOI":"10.1136\/ard-2023-224123","article-title":"The 2022 EULAR\/ACR points to consider at the early stages of diagnosis and management of suspected haemophagocytic lymphohistiocytosis\/macrophage activation syndrome (HLH\/MAS)","volume":"82","author":"Shakoory","year":"2023","journal-title":"Ann Rheum Dis"},{"key":"2025080201024117200_R5","doi-asserted-by":"crossref","first-page":"169","DOI":"10.1080\/14397595.2021.1899565","article-title":"Macrophage activation syndrome in patients with adult-onset Still\u2019s disease under tocilizumab treatment: a single-center observational study","volume":"32","author":"Yamabe","year":"2022","journal-title":"Mod Rheumatol"},{"key":"2025080201024117200_R6","doi-asserted-by":"crossref","first-page":"287","DOI":"10.1016\/j.cyto.2012.02.006","article-title":"Tocilizumab masks the clinical symptoms of systemic juvenile idiopathic arthritis-associated macrophage activation syndrome: the diagnostic significance of interleukin-18 and interleukin-6","volume":"58","author":"Shimizu","year":"2012","journal-title":"Cytokine"},{"key":"2025080201024117200_R7","doi-asserted-by":"crossref","first-page":"92","DOI":"10.3109\/s10165-010-0348-9","article-title":"Benefit and a possible risk of tocilizumab therapy for adult-onset Still\u2019s disease accompanied by macrophage-activation syndrome","volume":"21","author":"Kobayashi","year":"2011","journal-title":"Mod Rheumatol"},{"key":"2025080201024117200_R8","doi-asserted-by":"crossref","first-page":"1623","DOI":"10.1007\/s40744-023-00600-x","article-title":"Risk of macrophage activation syndrome in patients with adult-onset still\u2019s disease treated with IL-1 and IL-6 inhibitors: a meta-analysis and single-center experience","volume":"10","author":"Adachi","year":"2023","journal-title":"Rheumatol Ther"},{"key":"2025080201024117200_R9","first-page":"424","article-title":"Preliminary criteria for classification of adult Still\u2019s disease","volume":"19","author":"Yamaguchi","year":"1992","journal-title":"J Rheumatol"},{"key":"2025080201024117200_R10","doi-asserted-by":"crossref","first-page":"566","DOI":"10.1002\/art.39332","article-title":"Classification criteria for macrophage activation syndrome complicating systemic Juvenile idiopathic arthritis: a European league against rheumatism\/American College of Rheumatology\/Paediatric Rheumatology International trials organisation collaborative initiative","volume":"68","author":"Ravelli","year":"2016","journal-title":"Arthritis Rheumatol"},{"key":"2025080201024117200_R11","doi-asserted-by":"crossref","DOI":"10.1155\/2016\/5656320","article-title":"Successful tocilizumab therapy for macrophage 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serum gasdermin D N-terminal implicates monocyte and macrophage pyroptosis in adult-onset Still\u2019s disease","volume":"60","author":"Nagai","year":"2021","journal-title":"Rheumatology"},{"key":"2025080201024117200_R15","doi-asserted-by":"crossref","first-page":"1003","DOI":"10.1111\/1756-185X.14371","article-title":"Baseline clinical features predicting macrophage activation syndrome in patients with systemic adult-onset Still\u2019s disease receiving interleukin-6 inhibitor treatment","volume":"25","author":"Naniwa","year":"2022","journal-title":"Int J Rheum Dis"},{"key":"2025080201024117200_R16","doi-asserted-by":"crossref","first-page":"736","DOI":"10.1111\/1346-8138.16373","article-title":"Heliotrope-like manifestation of adult-onset Still disease with macrophage activation syndrome: a case-based review","volume":"49","author":"Ikeda","year":"2022","journal-title":"J Dermatol"},{"key":"2025080201024117200_R17","doi-asserted-by":"crossref","first-page":"360","DOI":"10.1080\/24725625.2020.1818961","article-title":"Reintroduction of tocilizumab elicited macrophage activation syndrome in a patient with adult-onset Still\u2019s disease with a previous successful tocilizumab treatment","volume":"5","author":"Naniwa","year":"2021","journal-title":"Mod Rheumatol Case Rep"},{"key":"2025080201024117200_R18","doi-asserted-by":"crossref","first-page":"553","DOI":"10.1002\/art.42037","article-title":"American College of Rheumatology guideline for the treatment of juvenile idiopathic arthritis: therapeutic approaches for oligoarthritis, temporomandibular joint arthritis, and systemic juvenile idiopathic arthritis","volume":"74","author":"Onel","year":"2021","journal-title":"Arthritis Rheumatol"},{"key":"2025080201024117200_R19","doi-asserted-by":"crossref","first-page":"174","DOI":"10.1093\/mr\/roae050","article-title":"Efficacy and safety of tumor necrosis 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