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This case describes a previously healthy mid-20s patient who was diagnosed with AOSD and subsequently MAS. Although his clinical symptoms and ferritin levels responded well to steroids, interleukin-1 inhibition and Janus kinase inhibition, he developed prolonged and profound neutropenia for which he received one dose of G-CSF. His MAS flared markedly, requiring intensive immunosuppression and significantly prolonging his hospital stay. This report illustrates that G-CSF carries the risk of worsening inflammation leading to MAS, particularly in auto-inflammatory conditions such as AOSD.<\/jats:p>","DOI":"10.1136\/bcr-2024-262024","type":"journal-article","created":{"date-parts":[[2025,2,24]],"date-time":"2025-02-24T23:55:30Z","timestamp":1740441330000},"page":"e262024","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":1,"title":["Marked exacerbation of resolving macrophage activation syndrome in a patient with adult-onset Still\u2019s disease following one dose of granulocyte colony-stimulating factor"],"prefix":"10.1136","volume":"18","author":[{"given":"Alana","family":"Petrassi","sequence":"first","affiliation":[{"name":"Department of Internal Medicine, University of Minnesota, Minneapolis, Minnesota, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Connor","family":"Buechler","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine, University of Minnesota, Minneapolis, Minnesota, USA"},{"name":"Department of Dermatology, University of Minnesota, Minneapolis, Minnesota, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2167-5894","authenticated-orcid":false,"given":"Priyanjali","family":"Pulipati","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine, University of Minnesota, Minneapolis, Minnesota, USA"},{"name":"Division of Rheumatic and Autoimmune Diseases, University of Minnesota Twin Cities, Minneapolis, Minnesota, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Elie","family":"Gertner","sequence":"additional","affiliation":[{"name":"Department of Internal Medicine, University of Minnesota, Minneapolis, Minnesota, USA"},{"name":"Division of Rheumatic and Autoimmune Diseases, University of Minnesota Twin Cities, Minneapolis, Minnesota, USA"},{"name":"Section of Rheumatology, Department of Internal Medicine, Regions Hospital, St Paul, Minnesota, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"239","published-online":{"date-parts":[[2025,2,24]]},"reference":[{"key":"2025082703211650000_18.2.e262024.1","doi-asserted-by":"publisher","DOI":"10.1159\/000312628"},{"key":"2025082703211650000_18.2.e262024.2","doi-asserted-by":"crossref","DOI":"10.3390\/jcm11010206","article-title":"Detection and Prediction of Macrophage Activation Syndrome in Still\u2019s Disease","volume":"11","author":"Javaux","year":"2021","journal-title":"J Clin Med"},{"key":"2025082703211650000_18.2.e262024.3","doi-asserted-by":"crossref","DOI":"10.3389\/fimmu.2022.955523","article-title":"Macrophage activation syndrome in adults: Characteristics, outcomes, and therapeutic effectiveness of etoposide-based regimen","volume":"13","author":"He","year":"2022","journal-title":"Front Immunol"},{"key":"2025082703211650000_18.2.e262024.4","doi-asserted-by":"crossref","DOI":"10.1016\/j.smim.2021.101516","article-title":"G-CSF - A double edge sword in neutrophil mediated immunity","volume":"54","author":"Martin","year":"2021","journal-title":"Semin Immunol"},{"key":"2025082703211650000_18.2.e262024.5","doi-asserted-by":"publisher","DOI":"10.3899\/jrheum.200617"},{"key":"2025082703211650000_18.2.e262024.6","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":"2025082703211650000_18.2.e262024.7","first-page":"194","article-title":"Proposal for a New Set of Classification Criteria for Adult-Onset Still Disease","volume":"81","author":"Fautrel","year":"2002","journal-title":"Medicine (Abingdon)"},{"key":"2025082703211650000_18.2.e262024.8","doi-asserted-by":"crossref","first-page":"e630","DOI":"10.1016\/S2352-3026(19)30156-5","article-title":"Ruxolitinib in adult patients with secondary haemophagocytic lymphohistiocytosis: an open-label, single-centre, pilot trial","volume":"6","author":"Ahmed","year":"2019","journal-title":"Lancet Haematol"},{"key":"2025082703211650000_18.2.e262024.9","doi-asserted-by":"publisher","DOI":"10.3324\/haematol.2019.222471"},{"key":"2025082703211650000_18.2.e262024.10","doi-asserted-by":"crossref","first-page":"797","DOI":"10.1007\/s10067-006-0238-x","article-title":"Hemophagocytic syndrome in one patient with adult-onset Still\u2019s disease. 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