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March 2022; 9 (2) Clinical/Scientific NoteOpen Access

Increased Intrathecal B and Plasma Cells in Patients With Anti-IgLON5 Disease

A Case Series

Christine Strippel, Anna Heidbreder, View ORCID ProfileAndreas Schulte-Mecklenbeck, Lisanne Korn, Tobias Warnecke, View ORCID ProfileNico Melzer, Heinz Wiendl, View ORCID ProfileMatthias Pawlowski, Catharina C. Gross, Stjepana Kovac
First published January 14, 2022, DOI: https://doi.org/10.1212/NXI.0000000000001137
Christine Strippel
From the Department of Neurology with Institute of Translational Neurology (C.S., A.S.-M., L.K., T.W., H.W., M.P., C.C.G., S.K.), University of Münster, Germany; Department of Neurology (A.H.), Medical University Innsbruck, Austria; and Department of Neurology (N.M.), Medical Faculty, Heinrich-Heine University of Düsseldorf, Germany.
MD
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  • For correspondence: christine.strippel@ukmuenster.de
Anna Heidbreder
From the Department of Neurology with Institute of Translational Neurology (C.S., A.S.-M., L.K., T.W., H.W., M.P., C.C.G., S.K.), University of Münster, Germany; Department of Neurology (A.H.), Medical University Innsbruck, Austria; and Department of Neurology (N.M.), Medical Faculty, Heinrich-Heine University of Düsseldorf, Germany.
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  • For correspondence: anna.heidbreder@i-med.ac.at
Andreas Schulte-Mecklenbeck
From the Department of Neurology with Institute of Translational Neurology (C.S., A.S.-M., L.K., T.W., H.W., M.P., C.C.G., S.K.), University of Münster, Germany; Department of Neurology (A.H.), Medical University Innsbruck, Austria; and Department of Neurology (N.M.), Medical Faculty, Heinrich-Heine University of Düsseldorf, Germany.
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  • ORCID record for Andreas Schulte-Mecklenbeck
  • For correspondence: andreas.schulte-mecklenbeck@ukmuenster.de
Lisanne Korn
From the Department of Neurology with Institute of Translational Neurology (C.S., A.S.-M., L.K., T.W., H.W., M.P., C.C.G., S.K.), University of Münster, Germany; Department of Neurology (A.H.), Medical University Innsbruck, Austria; and Department of Neurology (N.M.), Medical Faculty, Heinrich-Heine University of Düsseldorf, Germany.
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  • For correspondence: lisanne.korn@ukmuenster.de
Tobias Warnecke
From the Department of Neurology with Institute of Translational Neurology (C.S., A.S.-M., L.K., T.W., H.W., M.P., C.C.G., S.K.), University of Münster, Germany; Department of Neurology (A.H.), Medical University Innsbruck, Austria; and Department of Neurology (N.M.), Medical Faculty, Heinrich-Heine University of Düsseldorf, Germany.
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  • For correspondence: tobias.warnecke@ukmuenster.de
Nico Melzer
From the Department of Neurology with Institute of Translational Neurology (C.S., A.S.-M., L.K., T.W., H.W., M.P., C.C.G., S.K.), University of Münster, Germany; Department of Neurology (A.H.), Medical University Innsbruck, Austria; and Department of Neurology (N.M.), Medical Faculty, Heinrich-Heine University of Düsseldorf, Germany.
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  • For correspondence: nico.melzer@med.uni-duesseldorf.de
Heinz Wiendl
From the Department of Neurology with Institute of Translational Neurology (C.S., A.S.-M., L.K., T.W., H.W., M.P., C.C.G., S.K.), University of Münster, Germany; Department of Neurology (A.H.), Medical University Innsbruck, Austria; and Department of Neurology (N.M.), Medical Faculty, Heinrich-Heine University of Düsseldorf, Germany.
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  • For correspondence: heinz.wiendl@ukmuenster.de
Matthias Pawlowski
From the Department of Neurology with Institute of Translational Neurology (C.S., A.S.-M., L.K., T.W., H.W., M.P., C.C.G., S.K.), University of Münster, Germany; Department of Neurology (A.H.), Medical University Innsbruck, Austria; and Department of Neurology (N.M.), Medical Faculty, Heinrich-Heine University of Düsseldorf, Germany.
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Catharina C. Gross
From the Department of Neurology with Institute of Translational Neurology (C.S., A.S.-M., L.K., T.W., H.W., M.P., C.C.G., S.K.), University of Münster, Germany; Department of Neurology (A.H.), Medical University Innsbruck, Austria; and Department of Neurology (N.M.), Medical Faculty, Heinrich-Heine University of Düsseldorf, Germany.
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  • For correspondence: catharina.gross@ukmuenster.de
Stjepana Kovac
From the Department of Neurology with Institute of Translational Neurology (C.S., A.S.-M., L.K., T.W., H.W., M.P., C.C.G., S.K.), University of Münster, Germany; Department of Neurology (A.H.), Medical University Innsbruck, Austria; and Department of Neurology (N.M.), Medical Faculty, Heinrich-Heine University of Düsseldorf, Germany.
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Full PDF
Citation
Increased Intrathecal B and Plasma Cells in Patients With Anti-IgLON5 Disease
A Case Series
Christine Strippel, Anna Heidbreder, Andreas Schulte-Mecklenbeck, Lisanne Korn, Tobias Warnecke, Nico Melzer, Heinz Wiendl, Matthias Pawlowski, Catharina C. Gross, Stjepana Kovac
Neurol Neuroimmunol Neuroinflamm Mar 2022, 9 (2) 00; DOI: 10.1212/NXI.0000000000001137

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Abstract

Background and Objectives Despite detection of autoantibodies, anti-IgLON5 disease was historically considered a tau-associated neurodegenerative disease, with limited treatment options and detrimental consequences for the patients. Observations in increasing case numbers hint toward underlying inflammatory mechanisms that, early detection provided, open a valuable window of opportunity for therapeutic intervention. We aimed to further substantiate this view by studying the CSF of patients with anti-IgLON5.

Methods We identified 11 patients with anti-IgLON5 from our database and compared clinical, MRI, and CSF findings with a cohort of 20 patients with progressive supranuclear palsy (PSP) (as a noninflammatory tauopathy) and 22 patients with functional neurologic disorder.

Results Patients with anti-IgLON5 show inflammatory changes in routine CSF analysis, an increase in B-lymphocyte frequency, and the presence of plasma cells in comparison to the PSP-control group and functional neurologic disease controls. Patients with intrathecal plasma cells showed a clinical response to rituximab.

Discussion Our findings indicate the importance of inflammatory mechanisms, in particular in early and acute anti-IgLON5 cases, which may support the use of immune-suppressive treatments in these cases. The main limitation of the study is the small number of cases due to the rarity of the disease.

Footnotes

  • ↵* These authors contributed equally to this work as co–first authors.

  • ↵† These authors contributed equally to this work as co–senior authors.

  • Go to Neurology.org/NN for full disclosures. Funding information is provided at the end of the article.

  • The Article Processing Charge was funded by the authors.

  • Received August 23, 2021.
  • Accepted in final form December 3, 2021.
  • Copyright © 2022 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of the American Academy of Neurology.

This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivatives License 4.0 (CC BY-NC-ND), which permits downloading and sharing the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal.

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