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Temporal Dynamics of MOG Antibodies in Children With Acquired Demyelinating Syndrome

BACKGROUND AND OBJECTIVE: The spectrum of myelin oligodendrocyte glycoprotein (MOG) antibody–associated disorder (MOGAD) comprises monophasic diseases such as acute disseminated encephalomyelitis (ADEM), optic neuritis (ON), and transverse myelitis and relapsing courses of these presentations. Persi...

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Autores principales: Wendel, Eva Maria, Thonke, Helen Sophie, Bertolini, Annikki, Baumann, Matthias, Blaschek, Astrid, Merkenschlager, Andreas, Karenfort, Michael, Kornek, Barbara, Lechner, Christian, Pohl, Daniela, Pritsch, Martin, Schanda, Kathrin, Schimmel, Mareike, Thiels, Charlotte, Waltz, Stephan, Wiegand, Gert, Anlar, Banu, Barisic, Nina, Blank, Christian, Breu, Markus, Broser, Philip, Della Marina, Adela, Diepold, Katharina, Eckenweiler, Matthias, Eisenkölbl, Astrid, Freilinger, Michael, Gruber-Sedlmayr, Ursula, Hackenberg, Annette, Iff, Tobias, Knierim, Ellen, Koch, Johannes, Kutschke, Georg, Leiz, Steffen, Lischetzki, Grischa, Nosadini, Margherita, Pschibul, Alexander, Reiter-Fink, Edith, Rohrbach, Doris, Salandin, Michela, Sartori, Stefano, Schlump, Jan-Ulrich, Stoffels, Johannes, Strautmanis, Jurgis, Tibussek, Daniel, Tüngler, Victoria, Utzig, Norbert, Reindl, Markus, Rostásy, Kevin
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Lippincott Williams & Wilkins 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9562044/
https://www.ncbi.nlm.nih.gov/pubmed/36229191
http://dx.doi.org/10.1212/NXI.0000000000200035
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author Wendel, Eva Maria
Thonke, Helen Sophie
Bertolini, Annikki
Baumann, Matthias
Blaschek, Astrid
Merkenschlager, Andreas
Karenfort, Michael
Kornek, Barbara
Lechner, Christian
Pohl, Daniela
Pritsch, Martin
Schanda, Kathrin
Schimmel, Mareike
Thiels, Charlotte
Waltz, Stephan
Wiegand, Gert
Anlar, Banu
Barisic, Nina
Blank, Christian
Breu, Markus
Broser, Philip
Della Marina, Adela
Diepold, Katharina
Eckenweiler, Matthias
Eisenkölbl, Astrid
Freilinger, Michael
Gruber-Sedlmayr, Ursula
Hackenberg, Annette
Iff, Tobias
Knierim, Ellen
Koch, Johannes
Kutschke, Georg
Leiz, Steffen
Lischetzki, Grischa
Nosadini, Margherita
Pschibul, Alexander
Reiter-Fink, Edith
Rohrbach, Doris
Salandin, Michela
Sartori, Stefano
Schlump, Jan-Ulrich
Stoffels, Johannes
Strautmanis, Jurgis
Tibussek, Daniel
Tüngler, Victoria
Utzig, Norbert
Reindl, Markus
Rostásy, Kevin
author_facet Wendel, Eva Maria
Thonke, Helen Sophie
Bertolini, Annikki
Baumann, Matthias
Blaschek, Astrid
Merkenschlager, Andreas
Karenfort, Michael
Kornek, Barbara
Lechner, Christian
Pohl, Daniela
Pritsch, Martin
Schanda, Kathrin
Schimmel, Mareike
Thiels, Charlotte
Waltz, Stephan
Wiegand, Gert
Anlar, Banu
Barisic, Nina
Blank, Christian
Breu, Markus
Broser, Philip
Della Marina, Adela
Diepold, Katharina
Eckenweiler, Matthias
Eisenkölbl, Astrid
Freilinger, Michael
Gruber-Sedlmayr, Ursula
Hackenberg, Annette
Iff, Tobias
Knierim, Ellen
Koch, Johannes
Kutschke, Georg
Leiz, Steffen
Lischetzki, Grischa
Nosadini, Margherita
Pschibul, Alexander
Reiter-Fink, Edith
Rohrbach, Doris
Salandin, Michela
Sartori, Stefano
Schlump, Jan-Ulrich
Stoffels, Johannes
Strautmanis, Jurgis
Tibussek, Daniel
Tüngler, Victoria
Utzig, Norbert
Reindl, Markus
Rostásy, Kevin
author_sort Wendel, Eva Maria
collection PubMed
description BACKGROUND AND OBJECTIVE: The spectrum of myelin oligodendrocyte glycoprotein (MOG) antibody–associated disorder (MOGAD) comprises monophasic diseases such as acute disseminated encephalomyelitis (ADEM), optic neuritis (ON), and transverse myelitis and relapsing courses of these presentations. Persistently high MOG antibodies (MOG immunoglobulin G [IgG]) are found in patients with a relapsing disease course. Prognostic factors to determine the clinical course of children with a first MOGAD are still lacking. The objective of the study is to assess the clinical and laboratory prognostic parameters for a risk of relapse and the temporal dynamics of MOG‐IgG titers in children with MOGAD in correlation with clinical presentation and disease course. METHODS: In this prospective multicenter hospital-based study, children with a first demyelinating attack and complete data set comprising clinical and radiologic findings, MOG-IgG titer at onset, and clinical and serologic follow-up data were included. Serum samples were analyzed by live cell-based assay, and a titer level of ≥1:160 was classified as MOG-IgG–positive. RESULTS: One hundred sixteen children (f:m = 57:59) with MOGAD were included and initially diagnosed with ADEM (n = 59), unilateral ON (n = 12), bilateral ON (n = 16), myelitis (n = 6), neuromyelitis optica spectrum disorder (n = 8) or encephalitis (n = 6). The median follow-up time was 3 years in monophasic and 5 years in relapsing patients. There was no significant association between disease course and MOG-IgG titers at onset, sex, age at presentation, or clinical phenotype. Seroconversion to MOG-IgG–negative within 2 years of the initial event showed a significant risk reduction for a relapsing disease course. Forty-two/one hundred sixteen patients (monophasic n = 26, relapsing n = 16) had serial MOG-IgG testing in years 1 and 2 after the initial event. In contrast to relapsing patients, monophasic patients showed a significant decrease of MOG-IgG titers during the first and second years, often with seroconversion to negative titers. During the follow-up, MOG-IgG titers were persistently higher in relapsing than in monophasic patients. Decrease in MOG-IgG of ≥3 dilution steps after the first and second years was shown to be associated with a decreased risk of relapses. In our cohort, no patient experienced a relapse after seroconversion to MOG-IgG–negative. DISCUSSION: In this study, patients with declining MOG-IgG titers, particularly those with seroconversion to MOG-IgG–negative, are shown to have a significantly reduced relapse risk.
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spelling pubmed-95620442022-10-14 Temporal Dynamics of MOG Antibodies in Children With Acquired Demyelinating Syndrome Wendel, Eva Maria Thonke, Helen Sophie Bertolini, Annikki Baumann, Matthias Blaschek, Astrid Merkenschlager, Andreas Karenfort, Michael Kornek, Barbara Lechner, Christian Pohl, Daniela Pritsch, Martin Schanda, Kathrin Schimmel, Mareike Thiels, Charlotte Waltz, Stephan Wiegand, Gert Anlar, Banu Barisic, Nina Blank, Christian Breu, Markus Broser, Philip Della Marina, Adela Diepold, Katharina Eckenweiler, Matthias Eisenkölbl, Astrid Freilinger, Michael Gruber-Sedlmayr, Ursula Hackenberg, Annette Iff, Tobias Knierim, Ellen Koch, Johannes Kutschke, Georg Leiz, Steffen Lischetzki, Grischa Nosadini, Margherita Pschibul, Alexander Reiter-Fink, Edith Rohrbach, Doris Salandin, Michela Sartori, Stefano Schlump, Jan-Ulrich Stoffels, Johannes Strautmanis, Jurgis Tibussek, Daniel Tüngler, Victoria Utzig, Norbert Reindl, Markus Rostásy, Kevin Neurol Neuroimmunol Neuroinflamm Research Article BACKGROUND AND OBJECTIVE: The spectrum of myelin oligodendrocyte glycoprotein (MOG) antibody–associated disorder (MOGAD) comprises monophasic diseases such as acute disseminated encephalomyelitis (ADEM), optic neuritis (ON), and transverse myelitis and relapsing courses of these presentations. Persistently high MOG antibodies (MOG immunoglobulin G [IgG]) are found in patients with a relapsing disease course. Prognostic factors to determine the clinical course of children with a first MOGAD are still lacking. The objective of the study is to assess the clinical and laboratory prognostic parameters for a risk of relapse and the temporal dynamics of MOG‐IgG titers in children with MOGAD in correlation with clinical presentation and disease course. METHODS: In this prospective multicenter hospital-based study, children with a first demyelinating attack and complete data set comprising clinical and radiologic findings, MOG-IgG titer at onset, and clinical and serologic follow-up data were included. Serum samples were analyzed by live cell-based assay, and a titer level of ≥1:160 was classified as MOG-IgG–positive. RESULTS: One hundred sixteen children (f:m = 57:59) with MOGAD were included and initially diagnosed with ADEM (n = 59), unilateral ON (n = 12), bilateral ON (n = 16), myelitis (n = 6), neuromyelitis optica spectrum disorder (n = 8) or encephalitis (n = 6). The median follow-up time was 3 years in monophasic and 5 years in relapsing patients. There was no significant association between disease course and MOG-IgG titers at onset, sex, age at presentation, or clinical phenotype. Seroconversion to MOG-IgG–negative within 2 years of the initial event showed a significant risk reduction for a relapsing disease course. Forty-two/one hundred sixteen patients (monophasic n = 26, relapsing n = 16) had serial MOG-IgG testing in years 1 and 2 after the initial event. In contrast to relapsing patients, monophasic patients showed a significant decrease of MOG-IgG titers during the first and second years, often with seroconversion to negative titers. During the follow-up, MOG-IgG titers were persistently higher in relapsing than in monophasic patients. Decrease in MOG-IgG of ≥3 dilution steps after the first and second years was shown to be associated with a decreased risk of relapses. In our cohort, no patient experienced a relapse after seroconversion to MOG-IgG–negative. DISCUSSION: In this study, patients with declining MOG-IgG titers, particularly those with seroconversion to MOG-IgG–negative, are shown to have a significantly reduced relapse risk. Lippincott Williams & Wilkins 2022-10-13 /pmc/articles/PMC9562044/ /pubmed/36229191 http://dx.doi.org/10.1212/NXI.0000000000200035 Text en Copyright © 2022 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of the American Academy of Neurology. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-No Derivatives License 4.0 (CCBY-NC-ND) (https://creativecommons.org/licenses/by-nc-nd/4.0/) , where it is permissible to download and share the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal.
spellingShingle Research Article
Wendel, Eva Maria
Thonke, Helen Sophie
Bertolini, Annikki
Baumann, Matthias
Blaschek, Astrid
Merkenschlager, Andreas
Karenfort, Michael
Kornek, Barbara
Lechner, Christian
Pohl, Daniela
Pritsch, Martin
Schanda, Kathrin
Schimmel, Mareike
Thiels, Charlotte
Waltz, Stephan
Wiegand, Gert
Anlar, Banu
Barisic, Nina
Blank, Christian
Breu, Markus
Broser, Philip
Della Marina, Adela
Diepold, Katharina
Eckenweiler, Matthias
Eisenkölbl, Astrid
Freilinger, Michael
Gruber-Sedlmayr, Ursula
Hackenberg, Annette
Iff, Tobias
Knierim, Ellen
Koch, Johannes
Kutschke, Georg
Leiz, Steffen
Lischetzki, Grischa
Nosadini, Margherita
Pschibul, Alexander
Reiter-Fink, Edith
Rohrbach, Doris
Salandin, Michela
Sartori, Stefano
Schlump, Jan-Ulrich
Stoffels, Johannes
Strautmanis, Jurgis
Tibussek, Daniel
Tüngler, Victoria
Utzig, Norbert
Reindl, Markus
Rostásy, Kevin
Temporal Dynamics of MOG Antibodies in Children With Acquired Demyelinating Syndrome
title Temporal Dynamics of MOG Antibodies in Children With Acquired Demyelinating Syndrome
title_full Temporal Dynamics of MOG Antibodies in Children With Acquired Demyelinating Syndrome
title_fullStr Temporal Dynamics of MOG Antibodies in Children With Acquired Demyelinating Syndrome
title_full_unstemmed Temporal Dynamics of MOG Antibodies in Children With Acquired Demyelinating Syndrome
title_short Temporal Dynamics of MOG Antibodies in Children With Acquired Demyelinating Syndrome
title_sort temporal dynamics of mog antibodies in children with acquired demyelinating syndrome
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9562044/
https://www.ncbi.nlm.nih.gov/pubmed/36229191
http://dx.doi.org/10.1212/NXI.0000000000200035
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