{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,13]],"date-time":"2026-10-13T19:32:50Z","timestamp":1791919970813,"version":"4.3.4"},"reference-count":41,"publisher":"SAGE Publications","license":[{"start":{"date-parts":[[2020,1,1]],"date-time":"2020-01-01T00:00:00Z","timestamp":1577836800000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Therapeutic Advances in Musculoskeletal"],"published-print":{"date-parts":[[2020,1]]},"abstract":"<jats:p>Macrophage activation syndrome (MAS) is a severe, potentially fatal complication of rheumatic diseases. This case demonstrates the significant challenges and therapeutic considerations in adult-onset Still\u2019s disease (AOSD) complicated with MAS at initial presentation, which will be discussed. MAS in our patient was refractory to the first-line therapy with high-dose corticosteroids, early administration of anakinra at a standard dosage and subsequent add-on treatments with cyclosporine A, IVIG, etoposides and tocilizumab. At 2\u2009months after presentation, the patient was still critically ill with clinical, laboratory and histological signs of an active uncontrolled MAS. Notably, adoption of anakinra at a high dosage finally induced remission. This case confirms that adjusted dosage of anakinra is an effective therapeutic strategy in a severe AOSD-related MAS. It is tempting to speculate that anakinra at a high dosage, if used earlier, would have significantly changed the course of the disease in our patient and could have led to earlier remission.<\/jats:p>","DOI":"10.1177\/1759720x20974858","type":"journal-article","created":{"date-parts":[[2020,11,24]],"date-time":"2020-11-24T04:55:59Z","timestamp":1606193759000},"update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":20,"title":["Efficacy of high-dose anakinra in refractory macrophage activation syndrome in adult-onset Still\u2019s disease: when dosage matters in overcoming secondary therapy resistance"],"prefix":"10.1177","volume":"12","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-9162-9717","authenticated-orcid":false,"given":"Sofia","family":"Ajeganova","sequence":"first","affiliation":[{"name":"Rheumatology Department, Clinical Sciences, Vrije Universiteit Brussel, Universitair Ziekenhuis, Brussels, 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