{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,24]],"date-time":"2026-07-24T07:24:51Z","timestamp":1784877891432,"version":"3.55.0"},"reference-count":37,"publisher":"Oxford University Press (OUP)","issue":"2","license":[{"start":{"date-parts":[[2022,7,25]],"date-time":"2022-07-25T00:00:00Z","timestamp":1658707200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"funder":[{"DOI":"10.13039\/501100002347","name":"Bundesministerium f\u00fcr Bildung und Forschung","doi-asserted-by":"crossref","award":["01GM1908"],"award-info":[{"award-number":["01GM1908"]}],"id":[{"id":"10.13039\/501100002347","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Investissements d\u00b4Avenir","award":["ANR-18-RHUS-0012-BETPSY"],"award-info":[{"award-number":["ANR-18-RHUS-0012-BETPSY"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,2,13]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Autoimmune encephalitis can be classified into antibody-defined subtypes, which can manifest with immunotherapy-responsive movement disorders sometimes mimicking non-inflammatory aetiologies. In the elderly, anti-LGI1 and contactin associated protein like 2 (CASPR2) antibody-associated diseases compose a relevant fraction of autoimmune encephalitis. Patients with LGI1 autoantibodies are known to present with limbic encephalitis and additionally faciobrachial dystonic seizures may occur. However, the clinical spectrum of CASPR2 autoantibody-associated disorders is more diverse including limbic encephalitis, Morvan\u2019s syndrome, peripheral nerve hyperexcitability syndrome, ataxia, pain and sleep disorders. Reports on unusual, sometimes isolated and immunotherapy-responsive movement disorders in CASPR2 autoantibody-associated syndromes have caused substantial concern regarding necessity of autoantibody testing in patients with movement disorders. Therefore, we aimed to systematically assess their prevalence and manifestation in patients with CASPR2 autoimmunity.<\/jats:p>\n               <jats:p>This international, retrospective cohort study included patients with CASPR2 autoimmunity from participating expert centres in Europe. Patients with ataxia and\/or movement disorders were analysed in detail using questionnaires and video recordings. We recruited a comparator group with anti-LGI1 encephalitis from the GENERATE network. Characteristics were compared according to serostatus.<\/jats:p>\n               <jats:p>We identified 164 patients with CASPR2 autoantibodies. Of these, 149 (90.8%) had only CASPR2 and 15 (9.1%) both CASPR2 and LGI1 autoantibodies. Compared to 105 patients with LGI1 encephalitis, patients with CASPR2 autoantibodies more often had movement disorders and\/or ataxia (35.6 versus 3.8%; P &amp;lt; 0.001). This was evident in all subgroups: ataxia 22.6 versus 0.0%, myoclonus 14.6 versus 0.0%, tremor 11.0 versus 1.9%, or combinations thereof 9.8 versus 0.0% (all P &amp;lt; 0.001). The small group of patients double-positive for LGI1\/CASPR2 autoantibodies (15\/164) significantly more frequently had myoclonus, tremor, \u2018mixed movement disorders\u2019, Morvan\u2019s syndrome and underlying tumours. We observed distinct movement disorders in CASPR2 autoimmunity (14.6%): episodic ataxia (6.7%), paroxysmal orthostatic segmental myoclonus of the legs (3.7%) and continuous segmental spinal myoclonus (4.3%). These occurred together with further associated symptoms or signs suggestive of CASPR2 autoimmunity. However, 2\/164 patients (1.2%) had isolated segmental spinal myoclonus.<\/jats:p>\n               <jats:p>Movement disorders and ataxia are highly prevalent in CASPR2 autoimmunity. Paroxysmal orthostatic segmental myoclonus of the legs is a novel albeit rare manifestation. Further distinct movement disorders include isolated and combined segmental spinal myoclonus and autoimmune episodic ataxia.<\/jats:p>","DOI":"10.1093\/brain\/awac276","type":"journal-article","created":{"date-parts":[[2022,7,25]],"date-time":"2022-07-25T08:48:44Z","timestamp":1658738924000},"page":"657-667","source":"Crossref","is-referenced-by-count":40,"title":["Distinct movement disorders in contactin-associated-protein-like-2 antibody-associated autoimmune encephalitis"],"prefix":"10.1093","volume":"146","author":[{"given":"Felix","family":"G\u00f6vert","sequence":"first","affiliation":[{"name":"Department of Neurology, Christian-Albrecht University of Kiel and University Medical Center Schleswig-Holstein , 24105 Kiel , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ligia","family":"Abrante","sequence":"additional","affiliation":[{"name":"Neuroimmunology, Institute of Clinical Chemistry, Christian-Albrecht University of Kiel and University Medical Center Schleswig-Holstein , 24105 Kiel , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jos","family":"Becktepe","sequence":"additional","affiliation":[{"name":"Department of Neurology, Christian-Albrecht University of Kiel and University Medical Center Schleswig-Holstein , 24105 Kiel , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bettina","family":"Balint","sequence":"additional","affiliation":[{"name":"Department of Clinical and Movement Neurosciences, UCL Queen Square Institute of Neurology, Queen Square , London WC1N 3BG , UK"},{"name":"Department of Neurology, University Hospital Heidelberg , 69120 Heidelberg , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christos","family":"Ganos","sequence":"additional","affiliation":[{"name":"Department of Neurology, Charit\u00e9 University Medicine Berlin , 10117 Berlin , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ulrich","family":"Hofstadt-van Oy","sequence":"additional","affiliation":[{"name":"Department of Neurology, Klinikum Westfalen , 44309 Dortmund , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christos","family":"Krogias","sequence":"additional","affiliation":[{"name":"Department of Neurology, St Josef Hospital, Ruhr University Bochum , 44791 Bochum , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"James","family":"Varley","sequence":"additional","affiliation":[{"name":"Oxford Autoimmune Neurology Group, Nuffield Department of Clinical Neurosciences, University of Oxford, John Radcliffe Hospital , Oxford OX3 9DU , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7667-9748","authenticated-orcid":false,"given":"Sarosh R","family":"Irani","sequence":"additional","affiliation":[{"name":"Oxford Autoimmune Neurology Group, Nuffield Department of Clinical Neurosciences, University of Oxford, John Radcliffe Hospital , Oxford OX3 9DU , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sofija","family":"Paneva","sequence":"additional","affiliation":[{"name":"Oxford Autoimmune Neurology Group, Nuffield Department of Clinical Neurosciences, University of Oxford, John Radcliffe Hospital , Oxford OX3 9DU , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maarten J","family":"Titulaer","sequence":"additional","affiliation":[{"name":"Department of Neurology, Erasmus MC University Medical Center , 3015 GD Rotterdam , The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Juna M","family":"de Vries","sequence":"additional","affiliation":[{"name":"Department of Neurology, Erasmus MC University Medical Center , 3015 GD Rotterdam , The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Agnita J W","family":"Boon","sequence":"additional","affiliation":[{"name":"Department of Neurology, Erasmus MC University Medical Center , 3015 GD Rotterdam , The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marco W J","family":"Schreurs","sequence":"additional","affiliation":[{"name":"Department of Neurology, Erasmus MC University Medical Center , 3015 GD Rotterdam , The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bastien","family":"Joubert","sequence":"additional","affiliation":[{"name":"Centre National de R\u00e9f\u00e9rence pour les Syndromes Neurologiques Paran\u00e9oplasiques, Hospices Civils de Lyon, H\u00f4pital Neurologique , 69677 Bron , France"},{"name":"Institut NeuroMyoGene INSERM U1217\/CNRS UMR 5310, Universit\u00e9 de Lyon\u2013Universit\u00e9 Claude Bernard Lyon 1 , Lyon , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4721-5952","authenticated-orcid":false,"given":"Jerome","family":"Honnorat","sequence":"additional","affiliation":[{"name":"Centre National de R\u00e9f\u00e9rence pour les Syndromes Neurologiques Paran\u00e9oplasiques, Hospices Civils de Lyon, H\u00f4pital Neurologique , 69677 Bron , France"},{"name":"Institut NeuroMyoGene INSERM U1217\/CNRS UMR 5310, Universit\u00e9 de Lyon\u2013Universit\u00e9 Claude Bernard Lyon 1 , Lyon , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3652-7061","authenticated-orcid":false,"given":"Alberto","family":"Vogrig","sequence":"additional","affiliation":[{"name":"Centre National de R\u00e9f\u00e9rence pour les Syndromes Neurologiques Paran\u00e9oplasiques, Hospices Civils de Lyon, H\u00f4pital Neurologique , 69677 Bron , France"},{"name":"Institut NeuroMyoGene INSERM U1217\/CNRS UMR 5310, Universit\u00e9 de Lyon\u2013Universit\u00e9 Claude Bernard Lyon 1 , Lyon , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Helena","family":"Ari\u00f1o","sequence":"additional","affiliation":[{"name":"August Pi i Sunyer Biomedical Research Institute (IDIBAPS); Service of Neurology, Hospital Cl\u00ednic, University of Barcelona , 08036 Barcelona , Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lidia","family":"Sabater","sequence":"additional","affiliation":[{"name":"August Pi i Sunyer Biomedical Research Institute (IDIBAPS); Service of Neurology, Hospital Cl\u00ednic, University of Barcelona , 08036 Barcelona , Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Josep","family":"Dalmau","sequence":"additional","affiliation":[{"name":"August Pi i Sunyer Biomedical Research Institute (IDIBAPS); Service of Neurology, Hospital Cl\u00ednic, University of Barcelona , 08036 Barcelona , Spain"},{"name":"Department of Neurology, University of Pennsylvania , Philadelphia, PA 19104 , USA"},{"name":"Catalan Institution for Research and Advanced Studies (ICREA) , 08010 Barcelona , Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sangeeta","family":"Scotton","sequence":"additional","affiliation":[{"name":"Department of Neurology, University Hospitals Birmingham , Birmingham B15 2TH , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0037-454X","authenticated-orcid":false,"given":"Saiju","family":"Jacob","sequence":"additional","affiliation":[{"name":"Department of Neurology, University Hospitals Birmingham , Birmingham B15 2TH , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2420-701X","authenticated-orcid":false,"given":"Nico","family":"Melzer","sequence":"additional","affiliation":[{"name":"Department of Neurology with Institute of Translational Neurology, University of M\u00fcnster , 48149 M\u00fcnster , Germany"},{"name":"Department of Neurology, Medical Faculty, Heinrich-Heine-University D\u00fcsseldorf , 40225 D\u00fcsseldorf , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2225-8654","authenticated-orcid":false,"given":"Christian G","family":"Bien","sequence":"additional","affiliation":[{"name":"Department of Epileptology (Krankenhaus Mara), Bielefeld University, Medical School, Campus Bielefeld-Bethel , 33617 Bielefeld , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christian","family":"Geis","sequence":"additional","affiliation":[{"name":"Department of Neurology, University of Jena , 07747 Jena , Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9272-529X","authenticated-orcid":false,"given":"Jan","family":"Lewerenz","sequence":"additional","affiliation":[{"name":"Department of Neurology, Ulm University , 89081 Ulm , 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