{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,6]],"date-time":"2026-01-06T06:46:40Z","timestamp":1767682000534,"version":"3.48.0"},"reference-count":10,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2026,1,6]],"date-time":"2026-01-06T00:00:00Z","timestamp":1767657600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["frontiersin.org"],"crossmark-restriction":true},"short-container-title":["Front. Med."],"abstract":"<jats:p>While biologic monotherapy is commonly used in the management of moderate-to-severe systemic lupus erythematosus (SLE), limited data exists regarding the efficacy and safety of dual biologic therapy in SLE. We report the case of a 42-year-old woman with a history of SLE complicated by Rowell Syndrome (RS) that was managed on anifrolumab and a recent diagnosis of moyamoya disease, who presented with new-onset lower extremity weakness. Laboratory testing was notable for active lupus serologies, and magnetic resonance angiography (MRA) findings were consistent with worsening moyamoya syndrome. Given the clinical and radiographic findings, a diagnosis of CNS lupus was suspected. Anifrolumab was discontinued and she was started on pulse dose corticosteroids and rituximab, resulting in marked neurologic improvement. However, she subsequently experienced a flare of RS. Anifrolumab was reintroduced while she remained on rituximab, leading to significant clinical improvement of her cutaneous symptoms. To our knowledge, we present the first case of successful treatment of refractory SLE with both cutaneous and neurologic involvement utilizing dual biologic therapy with rituximab and anifrolumab.<\/jats:p>","DOI":"10.3389\/fmed.2025.1727404","type":"journal-article","created":{"date-parts":[[2026,1,6]],"date-time":"2026-01-06T06:41:19Z","timestamp":1767681679000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":0,"title":["Case Report: Relapsing systemic lupus erythematosus treated with dual rituximab and anifrolumab therapy"],"prefix":"10.3389","volume":"12","author":[{"given":"Sunnie","family":"Lee","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Janet","family":"Choi","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Steven","family":"Benitez","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jeanie","family":"Lee","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"1965","published-online":{"date-parts":[[2026,1,6]]},"reference":[{"key":"ref1","doi-asserted-by":"publisher","first-page":"343","DOI":"10.1046\/j.1365-2133.2000.03306.x","article-title":"Redefining Rowell's syndrome","volume":"142","author":"Zeitouni","year":"2000","journal-title":"Br J Dermatol"},{"key":"ref2","doi-asserted-by":"publisher","first-page":"501845","DOI":"10.1016\/j.reumae.2025.501845","article-title":"Use of anifrolumab in a patient with chronic multirefractory Rowell's syndrome","volume":"21","author":"Navarro-Joven","year":"2025","journal-title":"Reumatol Clin (Engl Ed)"},{"key":"ref3","doi-asserted-by":"publisher","first-page":"27","DOI":"10.1016\/j.jdcr.2022.11.008","article-title":"A case of Rowell syndrome with excellent improvement following anifrolumab","volume":"31","author":"Shope","year":"2022","journal-title":"JAAD Case Rep"},{"key":"ref4","doi-asserted-by":"publisher","first-page":"50","DOI":"10.1016\/j.jdcr.2023.12.020","article-title":"An impressive case of Rowell syndrome with extensive mucosal involvement successfully treated with anifrolumab","volume":"45","author":"Antohi","year":"2024","journal-title":"JAAD Case Rep"},{"key":"ref5","doi-asserted-by":"publisher","first-page":"971","DOI":"10.1111\/j.1365-2133.2012.10825.x","article-title":"The interferon-regulated gene signature is elevated in subacute cutaneous lupus erythematosus and discoid lupus erythematosus and correlates with the cutaneous lupus area and severity index score","volume":"166","author":"Braunstein","year":"2012","journal-title":"Br J Dermatol"},{"key":"ref6","doi-asserted-by":"publisher","first-page":"rkad077","DOI":"10.1093\/rap\/rkad077","article-title":"Systemic lupus erythematosus presenting with moyamoya and Guillain\u2013Barr\u00e9 syndrome","volume":"7","author":"Basnet","year":"2023","journal-title":"Rheumatol Adv Pract"},{"key":"ref7","doi-asserted-by":"publisher","first-page":"279","DOI":"10.1159\/000354109","article-title":"Moyamoya syndrome as an unusual presenting manifestation of systemic lupus erythematosus in a young woman","volume":"23","author":"Zhou","year":"2014","journal-title":"Med Princ Pract"},{"key":"ref8","doi-asserted-by":"publisher","first-page":"470","DOI":"10.1136\/ard.2006.057885","article-title":"Efficacy of rituximab (anti-CD20) for refractory systemic lupus erythematosus involving the central nervous system","volume":"66","author":"Tokunaga","year":"2007","journal-title":"Ann Rheum Dis"},{"key":"ref9","doi-asserted-by":"publisher","first-page":"2332","DOI":"10.1016\/S0140-6736(19)30237-5","article-title":"Management strategies and future directions for systemic lupus erythematosus in adults","volume":"393","author":"Durcan","year":"2019","journal-title":"Lancet"},{"key":"ref10","doi-asserted-by":"publisher","first-page":"103837","DOI":"10.1016\/j.autrev.2025.103837","article-title":"Dual B-cell targeting in systemic lupus erythematosus: the role of combined and sequential therapy with rituximab and belimumab","volume":"24","author":"Frade-Sosa","year":"2025","journal-title":"Autoimmun Rev"}],"container-title":["Frontiers in Medicine"],"original-title":[],"link":[{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2025.1727404\/full","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,1,6]],"date-time":"2026-01-06T06:41:42Z","timestamp":1767681702000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.frontiersin.org\/articles\/10.3389\/fmed.2025.1727404\/full"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,1,6]]},"references-count":10,"alternative-id":["10.3389\/fmed.2025.1727404"],"URL":"https:\/\/doi.org\/10.3389\/fmed.2025.1727404","relation":{},"ISSN":["2296-858X"],"issn-type":[{"type":"electronic","value":"2296-858X"}],"subject":[],"published":{"date-parts":[[2026,1,6]]},"article-number":"1727404"}}