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To investigate clinical and immunological effects of anti-CD19 CAR T cell use in treatment-refractory SPS, a 69-y-old female with a 9-y history of treatment-refractory SPS with deteriorating episodes of stiffness received an infusion of autologous anti-CD19 CAR T cells (KYV-101) and was monitored clinically and immunologically for more than 6 mo. CAR T cell infusion resulted in reduced leg stiffness, drastic improvement in gait, walking speed increase over 100%, and daily walking distance improvement from less than 50 m to over 6 km within 3 mo. GABAergic medication (benzodiazepines) was reduced by 40%. KYV-101 CAR T cells were well tolerated with only low-grade cytokine release syndrome. This report of successful use of anti-CD19 CAR T cells in treatment-refractory SPS supports continued exploration of this approach in SPS and other B cell\u2013related autoimmune disorders.<\/jats:p>","DOI":"10.1073\/pnas.2403227121","type":"journal-article","created":{"date-parts":[[2024,6,17]],"date-time":"2024-06-17T15:03:33Z","timestamp":1718636613000},"update-policy":"https:\/\/doi.org\/10.1073\/pnas.cm10313","source":"Crossref","is-referenced-by-count":83,"title":["Successful use of anti-CD19 CAR T cells in severe treatment-refractory stiff-person syndrome"],"prefix":"10.1073","volume":"121","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-3412-762X","authenticated-orcid":false,"given":"Simon","family":"Faissner","sequence":"first","affiliation":[{"name":"Department of Neurology, Ruhr-University Bochum, St. Josef-Hospital, Bochum 44791, 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