{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,6]],"date-time":"2026-10-06T06:50:36Z","timestamp":1791269436679,"version":"4.1.0"},"reference-count":31,"publisher":"Wiley","issue":"7","license":[{"start":{"date-parts":[[2019,3,25]],"date-time":"2019-03-25T00:00:00Z","timestamp":1553472000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Euro J of Neurology"],"published-print":{"date-parts":[[2019,7]]},"abstract":"<jats:sec><jats:title>Background and purpose<\/jats:title><jats:p>Acquired neuromyotonia can occur in patients with thymoma, alone or in association with myasthenia gravis (<jats:styled-content style=\"fixed-case\">MG<\/jats:styled-content>), but the clinical prognostic significance of such comorbidity is largely unknown. The clinico\u2010pathological features were investigated along with the occurrence of neuromyotonia as predictors of tumour recurrence in patients with thymoma\u2010associated myasthenia.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>A total number of 268 patients with thymomatous <jats:styled-content style=\"fixed-case\">MG<\/jats:styled-content> were studied retrospectively. Patients with symptoms of spontaneous muscle overactivity were selected for autoantibody testing using immunohistology for neuronal cell\u2010surface proteins and cell\u2010based assays for contactin\u2010associated protein 2 (<jats:styled-content style=\"fixed-case\">CASPR<\/jats:styled-content>2), leucine\u2010rich glioma inactivated 1 (<jats:styled-content style=\"fixed-case\">LGI<\/jats:styled-content>1), glycine receptor and Netrin\u20101 receptor antibodies. Neuromyotonia was diagnosed according to the presence of typical electromyography abnormalities and\/or autoantibodies against <jats:styled-content style=\"fixed-case\">LGI<\/jats:styled-content>1\/<jats:styled-content style=\"fixed-case\">CASPR<\/jats:styled-content>2.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Overall, 33\/268 (12%) <jats:styled-content style=\"fixed-case\">MG<\/jats:styled-content> patients had a thymoma recurrence. Five\/268 (2%) had neuromyotonia, four with typical autoantibodies, including <jats:styled-content style=\"fixed-case\">LGI<\/jats:styled-content>1 (<jats:italic>n<\/jats:italic>\u00a0=\u00a01), <jats:styled-content style=\"fixed-case\">CASPR<\/jats:styled-content>2 (<jats:italic>n<\/jats:italic>\u00a0=\u00a01) or both (<jats:italic>n<\/jats:italic>\u00a0=\u00a02). Three patients had Netrin\u20101 receptor antibodies, two with neuromyotonia and concomitant CASPR2+<jats:styled-content style=\"fixed-case\">LGI<\/jats:styled-content>1 antibodies and one with spontaneous muscle overactivity without electromyography evidence of neuromyotonia. Thymoma recurrence was more frequent in those with (4\/5, 80%) than in those without (28\/263, 10%, <jats:italic>P<\/jats:italic>\u00a0&lt;\u00a00.001) neuromyotonia. Neuromyotonia preceded the recurrence in 4\/5 patients. In univariate analysis, predictors of thymoma recurrence were age at thymectomy [odds ratio (<jats:styled-content style=\"fixed-case\">OR<\/jats:styled-content>) 0.95, 95% confidence interval (<jats:styled-content style=\"fixed-case\">CI<\/jats:styled-content>) 0.93\u20130.97], Masaoka stage \u2265<jats:styled-content style=\"fixed-case\">II<\/jats:styled-content>b (<jats:styled-content style=\"fixed-case\">OR<\/jats:styled-content> 10.73, 95% <jats:styled-content style=\"fixed-case\">CI<\/jats:styled-content> 2.38\u201348.36) and neuromyotonia (<jats:styled-content style=\"fixed-case\">OR<\/jats:styled-content> 41.78, 95% <jats:styled-content style=\"fixed-case\">CI<\/jats:styled-content> 4.71\u2013370.58).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p><jats:italic>De novo<\/jats:italic> occurrence of neuromyotonia in <jats:styled-content style=\"fixed-case\">MG<\/jats:styled-content> patients with previous thymomas is a rare event and may herald tumour recurrence. Neuronal autoantibodies can be helpful to assess the diagnosis. These observations provide pragmatic risk stratification for tumour vigilance in patients with thymomatous <jats:styled-content style=\"fixed-case\">MG<\/jats:styled-content>.<\/jats:p><\/jats:sec>","DOI":"10.1111\/ene.13922","type":"journal-article","created":{"date-parts":[[2019,2,4]],"date-time":"2019-02-04T04:15:59Z","timestamp":1549253759000},"page":"992-999","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":31,"title":["Acquired neuromyotonia in thymoma\u2010associated myasthenia gravis: a clinical and serological study"],"prefix":"10.1111","volume":"26","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-2288-2000","authenticated-orcid":false,"given":"M.","family":"Gastaldi","sequence":"first","affiliation":[{"name":"Neuroimmunology Laboratory IRCCS Mondino Foundation Pavia Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A.","family":"De Rosa","sequence":"additional","affiliation":[{"name":"Neurology Unit Department of Clinical and Experimental Medicine University of Pisa Pisa Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M.","family":"Maestri","sequence":"additional","affiliation":[{"name":"Neurology Unit Department of Clinical and Experimental Medicine University of Pisa Pisa Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"E.","family":"Zardini","sequence":"additional","affiliation":[{"name":"Neuroimmunology Laboratory IRCCS Mondino Foundation Pavia Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"S.","family":"Scaranzin","sequence":"additional","affiliation":[{"name":"Neuroimmunology Laboratory IRCCS Mondino Foundation Pavia Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M.","family":"Guida","sequence":"additional","affiliation":[{"name":"Neurology Unit Department of Clinical and Experimental Medicine University of Pisa Pisa Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"P.","family":"Borrelli","sequence":"additional","affiliation":[{"name":"Unit of Biostatistics and Clinical Epidemiology University of Pavia Pavia Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"O. 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