{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,29]],"date-time":"2026-10-29T23:31:30Z","timestamp":1793316690798,"version":"4.5.0"},"reference-count":22,"publisher":"Springer Science and Business Media LLC","issue":"8","license":[{"start":{"date-parts":[[2019,3,23]],"date-time":"2019-03-23T00:00:00Z","timestamp":1553299200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2019,3,23]],"date-time":"2019-03-23T00:00:00Z","timestamp":1553299200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100006396","name":"Alexion Pharmaceuticals","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100006396","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Qual Life Res"],"published-print":{"date-parts":[[2019,8,15]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec>\n<jats:title>Purpose<\/jats:title>\n<jats:p>To evaluate the effect of eculizumab on perceived fatigue in patients with anti-acetylcholine receptor antibody-positive, refractory, generalized myasthenia gravis (MG) using the Quality of Life in Neurological Disorders (Neuro-QOL) Fatigue subscale, and to evaluate correlations between improvements in Neuro-QOL Fatigue and other clinical endpoints.<\/jats:p>\n<\/jats:sec><jats:sec>\n<jats:title>Methods<\/jats:title>\n<jats:p>Neuro-QOL Fatigue, MG Activities of Daily Living (MG-ADL), Quantitative MG (QMG), and the 15-item MG Quality of Life (MG-QOL15) scales were administered during the phase 3, randomized, placebo-controlled REGAIN study (eculizumab, <jats:italic>n<\/jats:italic>\u2009=\u200962; placebo, <jats:italic>n<\/jats:italic>\u2009=\u200963) and subsequent open-label extension (OLE). Data were analyzed using repeated-measures models. Correlations between changes in Neuro-QOL Fatigue and in MG-ADL, QMG, and MG-QOL15 scores were determined at REGAIN week 26.<\/jats:p>\n<\/jats:sec><jats:sec>\n<jats:title>Results<\/jats:title>\n<jats:p>At REGAIN week 26, eculizumab-treated patients showed significantly greater improvements in Neuro-QOL Fatigue scores than placebo-treated patients (consistent with improvements in MG-ADL, QMG, and MG-QOL15 scores previously reported in REGAIN). Improvements with eculizumab were sustained through OLE week 52. Correlations between Neuro-QOL Fatigue and MG-QOL15, MG-ADL, and QMG scores were strong for eculizumab-treated patients at REGAIN week 26, and strong, moderate, and weak, respectively, for placebo-treated patients.<\/jats:p>\n<\/jats:sec><jats:sec>\n<jats:title>Conclusions<\/jats:title>\n<jats:p>Compared with placebo, eculizumab was associated with improvements in perceived fatigue that strongly correlated with improvements in MG-specific outcome measures.<\/jats:p>\n<jats:p><jats:bold>Trial ID Registration<\/jats:bold>: NCT01997229, NCT02301624.<\/jats:p>\n<\/jats:sec>","DOI":"10.1007\/s11136-019-02148-2","type":"journal-article","created":{"date-parts":[[2019,3,23]],"date-time":"2019-03-23T13:02:37Z","timestamp":1553346157000},"page":"2247-2254","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":49,"title":["Eculizumab improves fatigue in refractory generalized myasthenia 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Muscle & Nerve. https:\/\/doi.org\/10.1002\/mus.26447.","journal-title":"Muscle & Nerve"}],"updated-by":[{"DOI":"10.1007\/s11136-019-02204-x","type":"correction","label":"Correction","source":"publisher","updated":{"date-parts":[[2019,5,21]],"date-time":"2019-05-21T00:00:00Z","timestamp":1558396800000}}],"container-title":["Quality of Life Research"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s11136-019-02148-2.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1007\/s11136-019-02148-2\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s11136-019-02148-2.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,8,10]],"date-time":"2021-08-10T16:27:19Z","timestamp":1628612839000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/s11136-019-02148-2"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2019,3,23]]},"references-count":22,"journal-issue":{"issue":"8","published-print":{"date-parts":[[2019,8,15]]}},"alternative-id":["2148"],"URL":"https:\/\/doi.org\/10.1007\/s11136-019-02148-2","relation":{"correction":[{"id-type":"doi","id":"10.1007\/s11136-019-02204-x","asserted-by":"object"}]},"ISSN":["0962-9343","1573-2649"],"issn-type":[{"value":"0962-9343","type":"print"},{"value":"1573-2649","type":"electronic"}],"subject":[],"published":{"date-parts":[[2019,3,23]]},"assertion":[{"value":"21 February 2019","order":1,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"23 March 2019","order":2,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"21 May 2019","order":3,"name":"change_date","label":"Change Date","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"Correction","order":4,"name":"change_type","label":"Change Type","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"The article \u201cEculizumab improves fatigue in refractory generalized myasthenia gravis\u201d, written by \u201cHenning Andersen, Renato Mantegazza, Jing Jing Wang, Fanny O\u2019Brien, Kaushik Patra, James F. Howard Jr. and The REGAIN Study Group\u201d was originally published electronically on the publisher\u2019s internet portal (currently SpringerLink) on 23 March 2019 without open access.","order":5,"name":"change_details","label":"Change Details","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Compliance with Ethical Standards"}},{"value":"Henning Andersen, MD, DMSci, PhD, has received research and travel support, and speaker honoraria from CSL Behring, Eisai, Octapharma, Pfizer, Sanofi Genzyme and UCB Pharma and has served as a consultant on advisory boards for NMD Pharma, UCB Pharma and Sanofi Genzyme. Renato Mantegazza, MD, received funding for research and congress participation from Bayer, BioMarin, Sanofi Genzyme and Teva, and participated in Scientific Advisory Boards for Alexion Pharmaceuticals, Argenx BVBA and BioMarin. Jing Jing Wang, MD, was formerly employed by, and owns stock in, Alexion Pharmaceuticals. Fanny O\u2019Brien, PhD, and Kaushik Patra, PhD, are employees of, and own stock in, Alexion Pharmaceuticals. James F. Howard, Jr, MD, obtained: research support from Alexion Pharmaceuticals during the conduct of the study; grants from Alexion Pharmaceuticals; research support from Research Triangle Institute\/Centers for Disease Control and Prevention; grants from National Institutes of Health (including the National Institute of Neurological Disorders and Stroke, National Institute of Arthritis and Musculoskeletal and Skin Diseases); research support from the Muscular Dystrophy Association; honoraria from Alexion Pharmaceuticals, Argenx BVBA and Ra Pharmaceuticals; and non-financial support from Alexion Pharmaceuticals, Argenx BVBA, Ra Pharmaceuticals and Toleranzia AB, outside of the submitted work.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}},{"value":"This study was conducted in accordance with the ethical principles that have their origin in the Declaration of Helsinki, and are consistent with International Council for Harmonisation (ICH) harmonised tripartite guideline E6(R1): Good Clinical Practice (ICH E6[R1]) and applicable regulatory requirements.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Research involving human participants"}},{"value":"Written informed consent was obtained from all participants.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Informed consent"}}]}}