{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,3]],"date-time":"2026-09-03T15:29:13Z","timestamp":1788449353677,"version":"build-2803163510"},"reference-count":26,"publisher":"SAGE Publications","license":[{"start":{"date-parts":[[2025,1,1]],"date-time":"2025-01-01T00:00:00Z","timestamp":1735689600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2025,1,1]],"date-time":"2025-01-01T00:00:00Z","timestamp":1735689600000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"funder":[{"DOI":"10.13039\/100018748","name":"Argenx","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100018748","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["Ther Adv Neurol Disord"],"published-print":{"date-parts":[[2025,1]]},"abstract":"<jats:sec>\n                    <jats:title>Background:<\/jats:title>\n                    <jats:p>The ADAPT trial demonstrated the benefit of efgartigimod, a neonatal Fc receptor (FcRn) inhibitor, in acetylcholine receptor antibody (AChRAb) positive patients with generalized myasthenia gravis (MG). Information regarding the benefits in those lacking pathogenic antibodies is sparse.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objectives:<\/jats:title>\n                    <jats:p>We aimed to investigate the safety and efficacy of efgartigimod in patients with double-seronegative (SN) generalized MG.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Design:<\/jats:title>\n                    <jats:p>An open-label 6-month prospective study, conducted at our center.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods:<\/jats:title>\n                    <jats:p>Patients aged at least 18\u2009years with clinical and electrodiagnostic features of MG and negative results for AChRAb and muscle-specific tyrosine kinase antibodies were included. Efgartigimod was administered weekly for 4\u2009weeks and then biweekly for 5\u2009months followed by an observation period. The primary endpoint was the change in MG impairment index (MGII) at 6\u2009months compared to baseline. Secondary endpoints include the change in MG activities of daily living (MG-ADL), other MG scores, overall responders, and early responders. The safety analysis included all patients who received at least one dose of efgartigimod.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results:<\/jats:title>\n                    <jats:p>\n                      We enrolled 30 patients with SN MG who were resistant to other treatments and had unacceptable MGII scores. The MGII decreased by 11.92\u2009points (\n                      <jats:italic toggle=\"yes\">p<\/jats:italic>\n                      \u2009&lt;\u20090.01) with efgartigimod treatment. The MG-ADL also improved. Seventy-two percent of patients were responders with 31% being early responders. Adverse events were reported in 83.3% of patients, and in 90.6%, they were mild. Headache was the most common, reported in 26.7%, followed by flu\/common cold in 20%, and urinary tract infection in 13.3%.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion:<\/jats:title>\n                    <jats:p>Efgartigimod was well tolerated and efficacious in patients with SN MG. Future randomized, placebo-controlled studies are needed.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Trial registration:<\/jats:title>\n                    <jats:p>\n                      This trial is registered at ClinicalTrials.gov (NCT 06587867), accessed via\n                      <jats:ext-link xmlns:xlink=\"http:\/\/www.w3.org\/1999\/xlink\" ext-link-type=\"uri\" xlink:href=\"https:\/\/clinicaltrials.gov\/study\/NCT06587867?locStr=Toronto,%20ON,%20Canada&amp;country=Canada&amp;state=Ontario&amp;city=Toronto&amp;cond=Myasthenia%20Gravis&amp;intr=efgartigimod&amp;rank=1\">https:\/\/clinicaltrials.gov\/study\/NCT06587867?locStr=Toronto,%20ON,%20Canada&amp;country=Canada&amp;state=Ontario&amp;city=Toronto&amp;cond=Myasthenia%20Gravis&amp;intr=efgartigimod&amp;rank=1<\/jats:ext-link>\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1177\/17562864251388019","type":"journal-article","created":{"date-parts":[[2025,10,31]],"date-time":"2025-10-31T12:44:18Z","timestamp":1761914658000},"update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":7,"title":["Open-label study of efgartigimod in seronegative myasthenia gravis"],"prefix":"10.1177","volume":"18","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-2262-2674","authenticated-orcid":false,"given":"Mohamed","family":"Khateb","sequence":"first","affiliation":[{"name":"Ellen and Martin Prosserman Centre for Neuromuscular Diseases, University Health Network, University of Toronto, Toronto, ON, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ajith","family":"Sivadasan","sequence":"additional","affiliation":[{"name":"Ellen and Martin Prosserman Centre for Neuromuscular Diseases, University Health Network, University of Toronto, Toronto, ON, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carolina","family":"Barnett-Tapia","sequence":"additional","affiliation":[{"name":"Ellen and Martin Prosserman Centre for Neuromuscular Diseases, University Health Network, University of Toronto, Toronto, ON, Canada"},{"name":"Institute of Health Policy, Management and Evaluation, University of Toronto, Toronto, ON, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lubna","family":"Daniyal","sequence":"additional","affiliation":[{"name":"Ellen and Martin Prosserman Centre for Neuromuscular Diseases, University Health Network, University of Toronto, Toronto, ON, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lahiru","family":"Fernando","sequence":"additional","affiliation":[{"name":"Ellen and Martin Prosserman Centre for Neuromuscular Diseases, University Health Network, University of Toronto, Toronto, ON, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shiyi","family":"Chen","sequence":"additional","affiliation":[{"name":"Biostatistics Department, University Health Network, Toronto, ON, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Leif Erik","family":"Lovblom","sequence":"additional","affiliation":[{"name":"Institute of Health Policy, Management and Evaluation, University of Toronto, Toronto, ON, Canada"},{"name":"Biostatistics Department, University Health Network, Toronto, ON, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hans","family":"Katzberg","sequence":"additional","affiliation":[{"name":"Ellen and Martin Prosserman Centre for Neuromuscular Diseases, University Health Network, University of Toronto, Toronto, ON, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vera","family":"Bril","sequence":"additional","affiliation":[{"name":"Ellen and Martin Prosserman Centre for Neuromuscular Diseases, University Health Network, 5EC-309, Toronto General Hospital, University of Toronto, 200 Elizabeth Street, Toronto, ON M5G 2C4, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2025,10,31]]},"reference":[{"key":"e_1_3_4_2_2","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp.48.12.1246"},{"issue":"4","key":"e_1_3_4_3_2","doi-asserted-by":"crossref","first-page":"933","DOI":"10.1212\/WNL.51.4.933-a","article-title":"Antibody to acetylcholine receptor in myasthenia gravis: prevalence, clinical correlates, and diagnostic value","volume":"51","author":"Lindstrom JM","year":"1998","unstructured":"Lindstrom JM, Seybold ME, Lennon VA, et al. 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