{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T04:53:00Z","timestamp":1787028780671,"version":"build-2736575974"},"reference-count":48,"publisher":"MDPI AG","issue":"2","license":[{"start":{"date-parts":[[2024,1,29]],"date-time":"2024-01-29T00:00:00Z","timestamp":1706486400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"name":"Italian Ministry of University and Research (MUR)"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Life"],"abstract":"<jats:p>Adult-onset Still\u2019s disease (AOSD) is a rare systemic inflammatory disorder. Diagnosis can take a long time, especially in the presence of confounding factors, and it is, to some extent, a process of exclusion. AOSD has life-threating complications ranging from asymptomatic to severe, such as macrophage activation syndrome (MAS), which is also referred to as hemophagocytic lymphohistocytosis (HLH). This condition is correlated with cytokine storm production and monocyte\/macrophage overactivation and typically occurs with rash, pyrexia, pancytopenia, hepatosplenomegaly and systemic involvement. Exitus occurs in approximately 10% of cases. For the treatment of MAS-HLH, the Histiocyte Society currently suggests high-dose corticosteroids, with the possible addition of cyclosporine A, anti-interleukin (IL)-1, or IL-6 biological drugs; the inclusion of etoposide is recommended for the most severe conditions. In all cases, a multidisciplinary collaboration involving the resources and expertise of several specialists (e.g., rheumatologist, infectiologist, critical care medicine specialist) is advised. Herein, we provide a detailed description of the clinical case of a previously healthy young woman in which MAS developed as a dramatic onset manifestation of AOSD and whose diagnosis posed a real clinical challenge; the condition was finally resolved by applying the HLH-94 protocol (i.e., etoposide in combination with dexamethasone).<\/jats:p>","DOI":"10.3390\/life14020195","type":"journal-article","created":{"date-parts":[[2024,1,29]],"date-time":"2024-01-29T12:25:01Z","timestamp":1706531101000},"page":"195","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["Unveiling the Mystery of Adult-Onset Still\u2019s Disease: A Compelling Case Report"],"prefix":"10.3390","volume":"14","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-7863-8192","authenticated-orcid":false,"given":"Daniele","family":"Sola","sequence":"first","affiliation":[{"name":"Department of Translational Medicine, Universit\u00e0 del Piemonte Orientale, 28100 Novara, Italy"},{"name":"Internal Medicine Unit, Maggiore della Carit\u00e0 Hospital, 28100 Novara, Italy"},{"name":"CAAD (Center for Autoimmune and Allergic Diseases), Universit\u00e0 del Piemonte Orientale, 28100 Novara, Italy"},{"name":"IRCAD (Interdisciplinary Research Center of Autoimmune Diseases), Universit\u00e0 del Piemonte Orientale, 28100 Novara, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3338-5416","authenticated-orcid":false,"given":"Carlo","family":"Smirne","sequence":"additional","affiliation":[{"name":"Department of Translational Medicine, Universit\u00e0 del Piemonte Orientale, 28100 Novara, Italy"},{"name":"Internal Medicine Unit, Maggiore della Carit\u00e0 Hospital, 28100 Novara, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Francesco","family":"Bruggi","sequence":"additional","affiliation":[{"name":"Department of Translational Medicine, Universit\u00e0 del Piemonte Orientale, 28100 Novara, Italy"},{"name":"Internal Medicine Unit, Maggiore della Carit\u00e0 Hospital, 28100 Novara, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chiara","family":"Bottino Sbaratta","sequence":"additional","affiliation":[{"name":"Department of Translational Medicine, Universit\u00e0 del Piemonte Orientale, 28100 Novara, Italy"},{"name":"Internal Medicine Unit, Maggiore della Carit\u00e0 Hospital, 28100 Novara, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aubin Cardin","family":"Tamen Njata","sequence":"additional","affiliation":[{"name":"Department of Translational Medicine, Universit\u00e0 del Piemonte Orientale, 28100 Novara, Italy"},{"name":"Internal Medicine Unit, Maggiore della Carit\u00e0 Hospital, 28100 Novara, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Guido","family":"Valente","sequence":"additional","affiliation":[{"name":"Department of Translational Medicine, Universit\u00e0 del Piemonte Orientale, 28100 Novara, Italy"},{"name":"Pathology Unit, Sant\u2019Andrea Hospital, 13100 Vercelli, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maria Cristina","family":"Pavanelli","sequence":"additional","affiliation":[{"name":"Pathology Unit, Sant\u2019Andrea Hospital, 13100 Vercelli, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rosetta","family":"Vitetta","sequence":"additional","affiliation":[{"name":"Rheumatology Unit, Sant\u2019Andrea Hospital, 13100 Vercelli, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1488-8736","authenticated-orcid":false,"given":"Mattia","family":"Bellan","sequence":"additional","affiliation":[{"name":"Department of Translational Medicine, Universit\u00e0 del Piemonte Orientale, 28100 Novara, Italy"},{"name":"Internal Medicine Unit, Maggiore della Carit\u00e0 Hospital, 28100 Novara, Italy"},{"name":"CAAD (Center for Autoimmune and Allergic Diseases), Universit\u00e0 del Piemonte Orientale, 28100 Novara, Italy"},{"name":"IRCAD (Interdisciplinary Research Center of Autoimmune Diseases), Universit\u00e0 del Piemonte Orientale, 28100 Novara, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lorenzo","family":"De Paoli","sequence":"additional","affiliation":[{"name":"Hematology Unit, Sant\u2019Andrea Hospital, 13100 Vercelli, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9740-0155","authenticated-orcid":false,"given":"Mario","family":"Pirisi","sequence":"additional","affiliation":[{"name":"Department of Translational Medicine, Universit\u00e0 del Piemonte Orientale, 28100 Novara, Italy"},{"name":"Internal Medicine Unit, Maggiore della Carit\u00e0 Hospital, 28100 Novara, Italy"},{"name":"CAAD (Center for Autoimmune and Allergic Diseases), Universit\u00e0 del Piemonte Orientale, 28100 Novara, Italy"},{"name":"IRCAD (Interdisciplinary Research Center of Autoimmune Diseases), Universit\u00e0 del Piemonte Orientale, 28100 Novara, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2024,1,29]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"858","DOI":"10.1016\/j.semarthrit.2021.06.004","article-title":"Adult-Onset Still\u2019s Disease in Focus: Clinical Manifestations, Diagnosis, Treatment, and Unmet Needs in the Era of Targeted Therapies","volume":"51","author":"Efthimiou","year":"2021","journal-title":"Semin. 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