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Impaired Brain Growth in Myelin Oligodendrocyte Glycoprotein Antibody–Associated Acute Disseminated Encephalomyelitis

BACKGROUND AND OBJECTIVES: Acute disseminated encephalomyelitis (ADEM) is the most common phenotype in pediatric myelin oligodendrocyte glycoprotein (MOG) antibody–associated disease. A previous study demonstrated impaired brain growth in ADEM. However, the effect of MOG antibodies on brain growth r...

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Autores principales: Bartels, Frederik, Baumgartner, Birgit, Aigner, Annette, Cooper, Graham, Blaschek, Astrid, Wendel, Eva Maria, Bertolini, Annikki, Karenfort, Michael, Baumann, Matthias, Cleaveland, Robert, Wegener-Panzer, Andreas, Leiz, Steffen, Salandin, Michela, Krieg, Peter, Reindl, Tobias, Reindl, Markus, Finke, Carsten, Rostásy, Kevin
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Lippincott Williams & Wilkins 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9909582/
https://www.ncbi.nlm.nih.gov/pubmed/36754833
http://dx.doi.org/10.1212/NXI.0000000000200066
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author Bartels, Frederik
Baumgartner, Birgit
Aigner, Annette
Cooper, Graham
Blaschek, Astrid
Wendel, Eva Maria
Bertolini, Annikki
Karenfort, Michael
Baumann, Matthias
Cleaveland, Robert
Wegener-Panzer, Andreas
Leiz, Steffen
Salandin, Michela
Krieg, Peter
Reindl, Tobias
Reindl, Markus
Finke, Carsten
Rostásy, Kevin
author_facet Bartels, Frederik
Baumgartner, Birgit
Aigner, Annette
Cooper, Graham
Blaschek, Astrid
Wendel, Eva Maria
Bertolini, Annikki
Karenfort, Michael
Baumann, Matthias
Cleaveland, Robert
Wegener-Panzer, Andreas
Leiz, Steffen
Salandin, Michela
Krieg, Peter
Reindl, Tobias
Reindl, Markus
Finke, Carsten
Rostásy, Kevin
author_sort Bartels, Frederik
collection PubMed
description BACKGROUND AND OBJECTIVES: Acute disseminated encephalomyelitis (ADEM) is the most common phenotype in pediatric myelin oligodendrocyte glycoprotein (MOG) antibody–associated disease. A previous study demonstrated impaired brain growth in ADEM. However, the effect of MOG antibodies on brain growth remains unknown. Here, we performed brain volume analyses in MOG-positive and MOG-negative ADEM at onset and over time. METHODS: In this observational cohort study, we included a total of 62 MRI scans from 24 patients with ADEM (54.2% female; median age 5 years), of which 16 (66.7%) were MOG positive. Patients were compared with healthy controls from the NIH pediatric MRI data repository and a matched local cohort. Mixed-effect models were applied to assess group differences and other relevant factors, including relapses. RESULTS: At baseline and before any steroid treatment, patients with ADEM, irrespective of MOG antibody status, showed reduced brain volume compared with matched controls (median [interquartile range] 1,741.9 cm3 [1,645.1–1,805.2] vs 1,810.4 cm(3) [1,786.5–1,836.2]). Longitudinal analysis revealed reduced brain growth for both MOG-positive and MOG-negative patients with ADEM. However, MOG-negative patients showed a stronger reduction (−138.3 cm(3) [95% CI −193.6 to −82.9]) than MOG-positive patients (−50.0 cm(3) [−126.5 to −5.2]), independent of age, sex, and treatment. Relapsing patients (all MOG positive) showed additional brain volume loss (−15.8 cm(3) [−68.9 to 37.3]). DISCUSSION: Patients with ADEM exhibit brain volume loss and failure of age-expected brain growth. Importantly, MOG-negative status was associated with a more pronounced brain volume loss compared with MOG-positive patients.
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spelling pubmed-99095822023-02-09 Impaired Brain Growth in Myelin Oligodendrocyte Glycoprotein Antibody–Associated Acute Disseminated Encephalomyelitis Bartels, Frederik Baumgartner, Birgit Aigner, Annette Cooper, Graham Blaschek, Astrid Wendel, Eva Maria Bertolini, Annikki Karenfort, Michael Baumann, Matthias Cleaveland, Robert Wegener-Panzer, Andreas Leiz, Steffen Salandin, Michela Krieg, Peter Reindl, Tobias Reindl, Markus Finke, Carsten Rostásy, Kevin Neurol Neuroimmunol Neuroinflamm Research Article BACKGROUND AND OBJECTIVES: Acute disseminated encephalomyelitis (ADEM) is the most common phenotype in pediatric myelin oligodendrocyte glycoprotein (MOG) antibody–associated disease. A previous study demonstrated impaired brain growth in ADEM. However, the effect of MOG antibodies on brain growth remains unknown. Here, we performed brain volume analyses in MOG-positive and MOG-negative ADEM at onset and over time. METHODS: In this observational cohort study, we included a total of 62 MRI scans from 24 patients with ADEM (54.2% female; median age 5 years), of which 16 (66.7%) were MOG positive. Patients were compared with healthy controls from the NIH pediatric MRI data repository and a matched local cohort. Mixed-effect models were applied to assess group differences and other relevant factors, including relapses. RESULTS: At baseline and before any steroid treatment, patients with ADEM, irrespective of MOG antibody status, showed reduced brain volume compared with matched controls (median [interquartile range] 1,741.9 cm3 [1,645.1–1,805.2] vs 1,810.4 cm(3) [1,786.5–1,836.2]). Longitudinal analysis revealed reduced brain growth for both MOG-positive and MOG-negative patients with ADEM. However, MOG-negative patients showed a stronger reduction (−138.3 cm(3) [95% CI −193.6 to −82.9]) than MOG-positive patients (−50.0 cm(3) [−126.5 to −5.2]), independent of age, sex, and treatment. Relapsing patients (all MOG positive) showed additional brain volume loss (−15.8 cm(3) [−68.9 to 37.3]). DISCUSSION: Patients with ADEM exhibit brain volume loss and failure of age-expected brain growth. Importantly, MOG-negative status was associated with a more pronounced brain volume loss compared with MOG-positive patients. Lippincott Williams & Wilkins 2023-02-08 /pmc/articles/PMC9909582/ /pubmed/36754833 http://dx.doi.org/10.1212/NXI.0000000000200066 Text en Copyright © 2023 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-NonCommercial-NoDerivatives License 4.0 (CC BY-NC-ND) (https://creativecommons.org/licenses/by-nc-nd/4.0/) , 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.
spellingShingle Research Article
Bartels, Frederik
Baumgartner, Birgit
Aigner, Annette
Cooper, Graham
Blaschek, Astrid
Wendel, Eva Maria
Bertolini, Annikki
Karenfort, Michael
Baumann, Matthias
Cleaveland, Robert
Wegener-Panzer, Andreas
Leiz, Steffen
Salandin, Michela
Krieg, Peter
Reindl, Tobias
Reindl, Markus
Finke, Carsten
Rostásy, Kevin
Impaired Brain Growth in Myelin Oligodendrocyte Glycoprotein Antibody–Associated Acute Disseminated Encephalomyelitis
title Impaired Brain Growth in Myelin Oligodendrocyte Glycoprotein Antibody–Associated Acute Disseminated Encephalomyelitis
title_full Impaired Brain Growth in Myelin Oligodendrocyte Glycoprotein Antibody–Associated Acute Disseminated Encephalomyelitis
title_fullStr Impaired Brain Growth in Myelin Oligodendrocyte Glycoprotein Antibody–Associated Acute Disseminated Encephalomyelitis
title_full_unstemmed Impaired Brain Growth in Myelin Oligodendrocyte Glycoprotein Antibody–Associated Acute Disseminated Encephalomyelitis
title_short Impaired Brain Growth in Myelin Oligodendrocyte Glycoprotein Antibody–Associated Acute Disseminated Encephalomyelitis
title_sort impaired brain growth in myelin oligodendrocyte glycoprotein antibody–associated acute disseminated encephalomyelitis
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9909582/
https://www.ncbi.nlm.nih.gov/pubmed/36754833
http://dx.doi.org/10.1212/NXI.0000000000200066
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