{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,17]],"date-time":"2026-09-17T05:37:49Z","timestamp":1789623469732,"version":"build-2803163510"},"reference-count":22,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2026,5,22]],"date-time":"2026-05-22T00:00:00Z","timestamp":1779408000000},"content-version":"vor","delay-in-days":21,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"},{"start":{"date-parts":[[2026,5,1]],"date-time":"2026-05-01T00:00:00Z","timestamp":1777593600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Euro J of Neurology"],"published-print":{"date-parts":[[2026,5]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Long\u2010term outcome data for stiff person spectrum disorder (SPSD) remain limited and sustained response to treatment varies.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>In this retrospective cohort study, we included patients with probable or definite SPSD (Mayo Clinic criteria), evaluated at Mayo Clinic (12\/1995\u201007\/2024) with \u2265\u20095\u2009years of follow\u2010up after diagnosis. Primary outcome measures included the modified Rankin scale (mRS) and use of gait aids.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Fifty\u2010four patients were included, 41 (76%) were female; median age at onset was 45\u2009years (16\u201378). Most were antibody\u2010positive (51 [94%]), with high\u2010titer (serum, \u2265\u200920\u2009nmol\/L) GAD65\u2010IgG in 45 (83%). Nine patients had a stiff\u2010limb presentation (17%). The median interval from onset to diagnosis was 2\u2009years (0\u201334), and median follow\u2010up was 9\u2009years (5\u201326). Most patients (46 [85%]) received immune therapy; 37 (82%) reported improvement. All patients had significant impairment (mRS \u2265\u20092) at nadir, with 31 (57%) losing functional independence (mRS \u2265\u20093) and 40 (75%) requiring bilateral gait assistance. The median time from onset to nadir was 6\u2009years (0\u201339). At last follow\u2010up, most (37 [69%]) were functionally independent (mRS \u2264\u20092), including 8 (15%) with minimal restrictions (mRS \u2264\u20091). Bilateral gait assistance was needed in 22 (41%). Among 40 patients with \u2265\u200910\u2009years of follow\u2010up, 22 (56%) had an mRS \u2264\u20092 and 25 (63%) required a gait aid at last follow\u2010up. Poor outcome in this population was associated with bilateral gait assistance at nadir and older age (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&lt;\u20090.05).\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Although over half of SPSD patients lose functional independence at disease nadir, many regain independence and achieve well\u2010controlled symptoms.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/ene.70609","type":"journal-article","created":{"date-parts":[[2026,5,22]],"date-time":"2026-05-22T13:34:43Z","timestamp":1779456883000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Long\u2010Term Outcomes in Stiff Person Spectrum Disorder"],"prefix":"10.1111","volume":"33","author":[{"given":"Georgios","family":"Mangioris","sequence":"first","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic College of Medicine  Rochester Minnesota USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andrew","family":"McKeon","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic College of Medicine  Rochester Minnesota USA"},{"name":"Department of Neurology Mayo Clinic College of Medicine  Rochester Minnesota USA"},{"name":"Center for Multiple Sclerosis &amp; Autoimmune Neurology Mayo Clinic College of Medicine  Rochester Minnesota USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James H.","family":"Bower","sequence":"additional","affiliation":[{"name":"Department of Neurology Mayo Clinic College of Medicine  Rochester Minnesota USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0751-8967","authenticated-orcid":false,"given":"Nisa","family":"Vorasoot","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic College of Medicine  Rochester Minnesota USA"},{"name":"Department of Neurology Mayo Clinic College of Medicine  Rochester Minnesota USA"},{"name":"Center for Multiple Sclerosis &amp; Autoimmune Neurology Mayo Clinic College of Medicine  Rochester Minnesota USA"},{"name":"Division of Neurology, Department of Medicine, Faculty of Medicine Khon Kaen University  Khon Kaen Thailand"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Divyanshu","family":"Dubey","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic College of Medicine  Rochester Minnesota USA"},{"name":"Department of Neurology Mayo Clinic College of Medicine  Rochester Minnesota USA"},{"name":"Center for Multiple Sclerosis &amp; Autoimmune Neurology Mayo Clinic College of Medicine  Rochester Minnesota USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eoin P.","family":"Flanagan","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic College of Medicine  Rochester Minnesota USA"},{"name":"Department of Neurology Mayo Clinic College of Medicine  Rochester Minnesota USA"},{"name":"Center for Multiple Sclerosis &amp; Autoimmune Neurology Mayo Clinic College of Medicine  Rochester Minnesota USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sean J.","family":"Pittock","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic College of Medicine  Rochester Minnesota USA"},{"name":"Department of Neurology Mayo Clinic College of Medicine  Rochester Minnesota USA"},{"name":"Center for Multiple Sclerosis &amp; Autoimmune Neurology Mayo Clinic College of Medicine  Rochester Minnesota USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1960-9065","authenticated-orcid":false,"given":"Anastasia","family":"Zekeridou","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic College of Medicine  Rochester Minnesota USA"},{"name":"Department of Neurology Mayo Clinic College of Medicine  Rochester Minnesota USA"},{"name":"Center for Multiple Sclerosis &amp; 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