Distinct movement disorders in contactin-associated-protein-like-2 antibody-associated autoimmune encephalitis
| Autor: | Felix Gövert, Ligia Abrante, Jos Becktepe, Bettina Balint, Christos Ganos, Ulrich Hofstadt-van Oy, Christos Krogias, James Varley, Sarosh R Irani, Sofija Paneva, Maarten J Titulaer, Juna M de Vries, Agnita J W Boon, Marco W J Schreurs, Bastien Joubert, Jerome Honnorat, Alberto Vogrig, Helena Ariño, Lidia Sabater, Josep Dalmau, Sangeeta Scotton, Saiju Jacob, Nico Melzer, Christian G Bien, Christian Geis, Jan Lewerenz, Harald Prüss, Klaus-Peter Wandinger, Günther Deuschl, Frank Leypoldt |
|---|---|
| Přispěvatelé: | Neurology |
| Jazyk: | angličtina |
| Rok vydání: | 2023 |
| Předmět: |
Myoclonus
CASPR2 etiology [Movement Disorders] metabolism [Contactins] neuronal autoantibodies autoimmune encephalitis Autoimmune Diseases of the Nervous System Potassium Channels Voltage-Gated Limbic Encephalitis Tremor Humans Encephalitis movement disorders Ataxia Neurology (clinical) ddc:610 metabolism [Intracellular Signaling Peptides and Proteins] Aged Retrospective Studies Autoantibodies |
| Zdroj: | Brain 146(2), 657–667 (2023). doi:10.1093/brain/awac276 Brain : a journal of neurology, 146(2), 657-667. Oxford University Press |
| ISSN: | 1460-2156 0006-8950 |
| DOI: | 10.1093/brain/awac276 |
| Popis: | Autoimmune encephalitis (AE) can be classified into antibody-defined subtypes, which can manifest with immunotherapy-responsive movement disorders sometimes mimicking non-inflammatory etiologies. In the elderly, anti-leucin-rich-glioma-inactivated-1 (LGI1) and contactin-associated-protein-like-2 (CASPR2) antibody-associated diseases compose a relevant fraction of AE. Patients with LGI1 autoantibodies are known to present with limbic encephalitis and additionally faciobrachial dystonic seizures (FBDS) may occur. However, the clinical spectrum of CASPR2 autoantibody-associated disorders is more diverse including limbic encephalitis, Morvan`s 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. This international, retrospective cohort study included patients with CASPR2 autoimmunity from participating expert centers in Europe. Patients with ataxia and/or movement disorders were analyzed in detail using questionnaires and video recordings. We recruited a comparator group with anti-leucin-rich-glioma-inactivated-1 (LGI1) encephalitis from the GENERATE network. Characteristics were compared according to serostatus. 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 |
| Databáze: | OpenAIRE |
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