{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,17]],"date-time":"2026-09-17T01:39:17Z","timestamp":1789609157684,"version":"build-2803163510"},"reference-count":50,"publisher":"SAGE Publications","license":[{"start":{"date-parts":[[2023,1,1]],"date-time":"2023-01-01T00:00:00Z","timestamp":1672531200000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Ther Adv Neurol Disord"],"published-print":{"date-parts":[[2023,1]]},"abstract":"<jats:sec><jats:title>Background:<\/jats:title><jats:p> Neuromyelitis optica spectrum disorder (NMOSD) is a rare relapsing autoimmune disease of the central nervous system, affecting mainly optic nerves and spinal cord. NMOSD pathophysiology is associated with anti-aquaporin-4 (AQP4) immunoglobulin G (IgG) autoantibodies. Rapid extracorporeal elimination of autoantibodies with apheresis techniques, such as immunoadsorption (IA), was proven to be an effective treatment of NMOSD attacks. Data on the long-term use of IA to prevent attacks or progression of NMOSD are lacking. <\/jats:p><\/jats:sec><jats:sec><jats:title>Objectives:<\/jats:title><jats:p> The aim of this study was to evaluate efficacy and safety of maintenance IA for preventing recurrence of NMOSD attacks in patients refractory to other immunotherapies. <\/jats:p><\/jats:sec><jats:sec><jats:title>Design:<\/jats:title><jats:p> Case study. <\/jats:p><\/jats:sec><jats:sec><jats:title>Methods:<\/jats:title><jats:p> Retrospective analysis of two female patients with severe NMOSD refractory to conventional immunotherapies was performed. Both patients had responded to tryptophan IA (Tr-IA) as attack therapy and subsequently were treated with biweekly maintenance Tr-IA. <\/jats:p><\/jats:sec><jats:sec><jats:title>Results:<\/jats:title><jats:p> Patient 1 (AQP4-IgG seropositive, age 42\u2009years) had 1.38 attacks of optic neuritis per year within 10.1\u2009years before commencing regular Tr-IA. With maintenance Tr-IA for 3.1\u2009years, one mild attack occurred, which was responsive to steroid pulse therapy. Expanded Disability Status Scale (EDSS) was stable at 5.0. Visual function score of the last eye improved from 3 to 1. Patient 2 (AQP4-IgG seronegative, age 43\u2009years) experienced 1.7 attacks per year, mainly acute myelitis and optic neuritis, during the period of 10.0\u2009years before the start of Tr-IA. During regular Tr-IA treatment, no further NMOSD attack occurred. The patient was clinically stable without any additional immunosuppressive treatment for 5.3\u2009years. EDSS improved from 6.0 to 5.0, and the ambulation score from 7 to 1. Tolerability of Tr-IA was good in both patients. No serious adverse events occurred during long-term clinical trajectories. <\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion:<\/jats:title><jats:p> Tr-IA was well tolerated as maintenance treatment and resulted in clinical stabilization of two patients with highly active NMOSD, who were refractory to standard drug therapy. <\/jats:p><\/jats:sec>","DOI":"10.1177\/17562864221150314","type":"journal-article","created":{"date-parts":[[2023,2,7]],"date-time":"2023-02-07T05:40:26Z","timestamp":1675748426000},"update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":8,"title":["Immunoadsorption as maintenance therapy for refractory neuromyelitis optica spectrum disorder"],"prefix":"10.1177","volume":"16","author":[{"given":"Franz","family":"Heigl","sequence":"first","affiliation":[{"name":"Medical Care Center Kempten-Allg\u00e4u, Kempten, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Reinhard","family":"Hettich","sequence":"additional","affiliation":[{"name":"Medical Care Center Kempten-Allg\u00e4u, Kempten, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6498-0210","authenticated-orcid":false,"given":"Cordula","family":"Fassbender","sequence":"additional","affiliation":[{"name":"Apheresis Research Institute, Cologne, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Reinhard","family":"Klingel","sequence":"additional","affiliation":[{"name":"Apheresis Research Institute, Cologne, Germany; 1st Department of Internal Medicine, University of Mainz, Mainz, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Erich","family":"Mauch","sequence":"additional","affiliation":[{"name":"Clinic for Neurology Dietenbronn, Academic Hospital of University of Ulm, Schwendi, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joachim","family":"Durner","sequence":"additional","affiliation":[{"name":"Neurology Department, M&I Fachklinik Ichenhausen, Ichenhausen, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rolf","family":"Kern","sequence":"additional","affiliation":[{"name":"Department of Neurology, Klinikum Worms, Worms, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ingo","family":"Kleiter","sequence":"additional","affiliation":[{"name":"Marianne-Strau\u00df-Klinik, Behandlungszentrum Kempfenhausen f\u00fcr Multiple Sklerose Kranke gGmbH, Milchberg 21, 82335 Berg, Germany"},{"name":"Department of Neurology, Ruhr-University Bochum, Bochum, Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2023,2,6]]},"reference":[{"key":"bibr1-17562864221150314","doi-asserted-by":"publisher","DOI":"10.1038\/s41572-020-0214-9"},{"key":"bibr2-17562864221150314","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000001729"},{"key":"bibr3-17562864221150314","doi-asserted-by":"publisher","DOI":"10.1186\/s12974-018-1144-2"},{"key":"bibr4-17562864221150314","doi-asserted-by":"publisher","DOI":"10.1016\/S1474-4422(21)00218-0"},{"key":"bibr5-17562864221150314","doi-asserted-by":"publisher","DOI":"10.1007\/s13311-015-0400-8"},{"key":"bibr6-17562864221150314","doi-asserted-by":"publisher","DOI":"10.1002\/jca.21705"},{"key":"bibr7-17562864221150314","unstructured":"Hemmer B. 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