{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,18]],"date-time":"2026-10-18T19:40:12Z","timestamp":1792352412073,"version":"4.4.0"},"reference-count":12,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2026,10,18]],"date-time":"2026-10-18T00:00:00Z","timestamp":1792281600000},"content-version":"vor","delay-in-days":290,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,1,1]],"date-time":"2026-01-01T00:00:00Z","timestamp":1767225600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Case Reports in Rheumatology"],"published-print":{"date-parts":[[2026,1]]},"abstract":"<jats:p>Hemophagocytic lymphohistiocytosis (HLH) is a life\u2010threatening, systemic inflammatory syndrome. Cases without an identifiable acquired trigger are referred to as secondary HLH. In this report, we describe the case of a 24 year\u2010old woman whose underlying cause of HLH was particularly difficult to determine. Her condition was initially suspected to be due to lamotrigine use but is now thought to be a result of undifferentiated connective tissue disease. During the patient\u2019s prolonged and complex hospital stay, she received the diagnosis of secondary HLH. Discontinuing lamotrigine, a drug that rarely causes HLH, did not improve her condition. The patient was tested for infectious and malignant conditions with negative results. Further workup revealed a positive ANA and bilateral MCP tenosynovitis, with negative specific antibodies, suggesting an undefined rheumatologic process. A number of conventional therapies did not show clear benefit, including etoposide, high\u2010dose steroids, rituximab, and plasmapheresis. The patient remained in critical condition until she began treatment with ruxolitinib, with which she remains stable more than 5\u2009years later. Ruxolitinib has shown some success in treating HLH in preliminary studies, but it is still considered a salvage therapy. This case illustrates the successful use of ruxolitinib in a patient with HLH likely due to undifferentiated connective tissue disease.<\/jats:p>","DOI":"10.1155\/crrh\/9165820","type":"journal-article","created":{"date-parts":[[2026,10,18]],"date-time":"2026-10-18T19:08:01Z","timestamp":1792350481000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Hemophagocytic Lymphohistiocytosis Thought to Be due to Undifferentiated Connective Tissue Disease Treated With Ruxolitinib: A Case Report"],"prefix":"10.1155","volume":"2026","author":[{"ORCID":"https:\/\/orcid.org\/0009-0002-3638-482X","authenticated-orcid":false,"given":"Emily","family":"Vachon","sequence":"first","affiliation":[{"name":"Carle Illinois College of Medicine  Urbana Illinois USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0002-8323-3530","authenticated-orcid":false,"given":"Heena Birbal","family":"Jain","sequence":"additional","affiliation":[{"name":"Carle Foundation Hospital  Urbana Illinois USA  carle.org"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0007-9808-2807","authenticated-orcid":false,"given":"Suzanna","family":"Kitten","sequence":"additional","affiliation":[{"name":"Carle Illinois College of Medicine  Urbana Illinois USA"},{"name":"Carle Foundation Hospital  Urbana Illinois USA  carle.org"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,10,18]]},"reference":[{"key":"e_1_2_9_1_2","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.21039"},{"key":"e_1_2_9_2_2","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2011-07-370148"},{"key":"e_1_2_9_3_2","volume-title":"GeneReviews\u00ae","author":"Zhang K.","year":"2006"},{"key":"e_1_2_9_4_2","doi-asserted-by":"publisher","DOI":"10.1186\/s12879-024-09356-3"},{"key":"e_1_2_9_5_2","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2016-12-753830"},{"key":"e_1_2_9_6_2","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000007517"},{"key":"e_1_2_9_7_2","first-page":"1414","article-title":"Haemophagocytic Syndrome in Rheumatic Patients. 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