Evolution of brain MRI lesions in paediatric myelin-oligodendrocyte glycoprotein antibody-associated disease (MOGAD) and its relevance to disease course

Omar Abdel-Mannan; Dimitrios Champsas; Carmen Tur; Vanessa Lee; Sharmila Manivannan; Haroon Usman; Alison Skippen; Ishita Desai; Manali Chitre; Rob Forsyth; Rachel Kneen; Dipak Ram; Sithara Ramdas; Thomas Rossor; Siobhan West; Sukhvir Wright; Jacqueline Palace; Evangeline Wassmer; Cheryl Hemingway; Ming J Lim; Kshitij Mankad; Olga Ciccarelli; Yael Hacohen

doi:10.1136/jnnp-2023-332542

Evolution of brain MRI lesions in paediatric myelin-oligodendrocyte glycoprotein antibody-associated disease (MOGAD) and its relevance to disease course

Omar Abdel-Mannan, Dimitrios Champsas, Carmen Tur, Vanessa Lee, Sharmila Manivannan, Haroon Usman, Alison Skippen, Ishita Desai, Manali Chitre, Rob Forsyth, Rachel Kneen, Dipak Ram, Sithara Ramdas, Thomas Rossor, Siobhan West, Sukhvir Wright, Jacqueline Palace, Evangeline Wassmer, Cheryl Hemingway, Ming J LimKshitij Mankad, Olga Ciccarelli, Yael Hacohen^*

^*Corresponding author for this work

Research output: Contribution to journal › Article › peer-review

Abstract

Background: Lesion resolution is often observed in children with myelin-oligodendrocyte glycoprotein antibody-associated disease (MOGAD), and asymptomatic lesions are less commonly reported in MOGAD than in multiple sclerosis (MS).

Objective: We aimed to evaluate brain MRI changes over time in paediatric MOGAD.

Methods: Retrospective study in eight UK paediatric neuroscience centres. Acute brain MRI and available follow-up MRIs were reviewed. Predictors for lesion dynamic were evaluated using multivariable regression and Kaplan-Meier survival analyses were used to predict risk of relapse, disability and MOG-Ab status.

Results:200 children were included (MOGAD 97; MS 103). At first MRI post-attack, new symptomatic and asymptomatic lesions were seen more often in MS vs MOGAD (52/103 vs 28/97; p=0.002 and 37/103 vs 11/97; p<0.001); 83% of MOGAD patients showed at least one lesion’s resolution at 1st follow‐up scan, and 23% had normal MRI. Only 1 MS patient had single lesion
resolution; none had normal MRI. Disappearing lesions in MOGAD were seen in 40% after the 2nd attack, 21% after 3rd attack and none after the 4th attack.

New lesions at 1st follow-up scan were associated with increased likelihood of relapse (p=0.02) and persistent MOG-Ab serostatus (p=0.0016) compared to those with no new lesions. Plasma exchange was associated with increased likelihood of lesion resolution (p=0.01). Longer time from symptom onset to steroids was associated with increased likelihood of new lesions; 50% increase at 20 days (p=0.01).

Conclusions and Relevance: These striking differences in lesion dynamics between MOGAD and MS suggest greater potential to repair. Early treatment with steroids and plasma exchange is associated with reduced likelihood of new lesions.

Original language	English
Article number	jnnp-2023-332542
Journal	Journal of Neurology, Neurosurgery and Psychiatry
Early online date	18 Nov 2023
DOIs	https://doi.org/10.1136/jnnp-2023-332542
Publication status	E-pub ahead of print - 18 Nov 2023

Bibliographical note

Copyright © Author(s), 2023. This article has been accepted for publication in the Journal of Neurology, Neurosurgery & Psychiatry following peer review, and the Version of Record can be accessed online at: https://doi.org/10.1136/jnnp-2023-332542. Reuse of this manuscript version (excluding any databases, tables, diagrams, photographs and other images or illustrative material included where a another copyright owner is identified) is permitted strictly pursuant to the terms of the Creative Commons Attribution-Non Commercial 4.0 International license (CC-BY-NC 4.0) https://creativecommons.org/licenses/by-nc/4.0/

Keywords

PAEDIATRIC NEUROLOGY
MYELIN
MRI
NEUROIMMUNOLOGY
MULTIPLE SCLEROSIS

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10.1136/jnnp-2023-332542

Abdel-Mannan et al_AAM_Evolution of brain MRI lesions in Paediatric MOGAD and relevance to disease course
Copyright © Author(s), 2023. This article has been accepted for publication in the Journal of Neurology, Neurosurgery & Psychiatry following peer review, and the Version of Record can be accessed online at: https://doi.org/10.1136/jnnp-2023-332542. Reuse of this manuscript version (excluding any databases, tables, diagrams, photographs and other images or illustrative material included where a another copyright owner is identified) is permitted strictly pursuant to the terms of the Creative Commons Attribution-Non Commercial 4.0 International license (CC-BY-NC 4.0) https://creativecommons.org/licenses/by-nc/4.0/
Accepted author manuscript, 792 KBLicence: CC BY-NC 4.0

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Abdel-Mannan, O., Champsas, D., Tur, C., Lee, V., Manivannan, S., Usman, H., Skippen, A., Desai, I., Chitre, M., Forsyth, R., Kneen, R., Ram, D., Ramdas, S., Rossor, T., West, S., Wright, S., Palace, J., Wassmer, E., Hemingway, C., ... Hacohen, Y. (2023). Evolution of brain MRI lesions in paediatric myelin-oligodendrocyte glycoprotein antibody-associated disease (MOGAD) and its relevance to disease course. Journal of Neurology, Neurosurgery and Psychiatry, Article jnnp-2023-332542. Advance online publication. https://doi.org/10.1136/jnnp-2023-332542

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abstract = "Background: Lesion resolution is often observed in children with myelin-oligodendrocyte glycoprotein antibody-associated disease (MOGAD), and asymptomatic lesions are less commonly reported in MOGAD than in multiple sclerosis (MS). Objective: We aimed to evaluate brain MRI changes over time in paediatric MOGAD. Methods: Retrospective study in eight UK paediatric neuroscience centres. Acute brain MRI and available follow-up MRIs were reviewed. Predictors for lesion dynamic were evaluated using multivariable regression and Kaplan-Meier survival analyses were used to predict risk of relapse, disability and MOG-Ab status. Results:200 children were included (MOGAD 97; MS 103). At first MRI post-attack, new symptomatic and asymptomatic lesions were seen more often in MS vs MOGAD (52/103 vs 28/97; p=0.002 and 37/103 vs 11/97; p<0.001); 83% of MOGAD patients showed at least one lesion{\textquoteright}s resolution at 1st follow‐up scan, and 23% had normal MRI. Only 1 MS patient had single lesionresolution; none had normal MRI. Disappearing lesions in MOGAD were seen in 40% after the 2nd attack, 21% after 3rd attack and none after the 4th attack.New lesions at 1st follow-up scan were associated with increased likelihood of relapse (p=0.02) and persistent MOG-Ab serostatus (p=0.0016) compared to those with no new lesions. Plasma exchange was associated with increased likelihood of lesion resolution (p=0.01). Longer time from symptom onset to steroids was associated with increased likelihood of new lesions; 50% increase at 20 days (p=0.01).Conclusions and Relevance: These striking differences in lesion dynamics between MOGAD and MS suggest greater potential to repair. Early treatment with steroids and plasma exchange is associated with reduced likelihood of new lesions.",

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note = "Copyright {\textcopyright} Author(s), 2023. This article has been accepted for publication in the Journal of Neurology, Neurosurgery & Psychiatry following peer review, and the Version of Record can be accessed online at: https://doi.org/10.1136/jnnp-2023-332542. Reuse of this manuscript version (excluding any databases, tables, diagrams, photographs and other images or illustrative material included where a another copyright owner is identified) is permitted strictly pursuant to the terms of the Creative Commons Attribution-Non Commercial 4.0 International license (CC-BY-NC 4.0) https://creativecommons.org/licenses/by-nc/4.0/",

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doi = "10.1136/jnnp-2023-332542",

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Abdel-Mannan, O, Champsas, D, Tur, C, Lee, V, Manivannan, S, Usman, H, Skippen, A, Desai, I, Chitre, M, Forsyth, R, Kneen, R, Ram, D, Ramdas, S, Rossor, T, West, S, Wright, S, Palace, J, Wassmer, E, Hemingway, C, Lim, MJ, Mankad, K, Ciccarelli, O & Hacohen, Y 2023, 'Evolution of brain MRI lesions in paediatric myelin-oligodendrocyte glycoprotein antibody-associated disease (MOGAD) and its relevance to disease course', Journal of Neurology, Neurosurgery and Psychiatry. https://doi.org/10.1136/jnnp-2023-332542

TY - JOUR

T1 - Evolution of brain MRI lesions in paediatric myelin-oligodendrocyte glycoprotein antibody-associated disease (MOGAD) and its relevance to disease course

AU - Abdel-Mannan, Omar

AU - Champsas, Dimitrios

AU - Tur, Carmen

AU - Lee, Vanessa

AU - Manivannan, Sharmila

AU - Usman, Haroon

AU - Skippen, Alison

AU - Desai, Ishita

AU - Chitre, Manali

AU - Forsyth, Rob

AU - Kneen, Rachel

AU - Ram, Dipak

AU - Ramdas, Sithara

AU - Rossor, Thomas

AU - West, Siobhan

AU - Wright, Sukhvir

AU - Palace, Jacqueline

AU - Wassmer, Evangeline

AU - Hemingway, Cheryl

AU - Lim, Ming J

AU - Mankad, Kshitij

AU - Ciccarelli, Olga

AU - Hacohen, Yael

N1 - Copyright © Author(s), 2023. This article has been accepted for publication in the Journal of Neurology, Neurosurgery & Psychiatry following peer review, and the Version of Record can be accessed online at: https://doi.org/10.1136/jnnp-2023-332542. Reuse of this manuscript version (excluding any databases, tables, diagrams, photographs and other images or illustrative material included where a another copyright owner is identified) is permitted strictly pursuant to the terms of the Creative Commons Attribution-Non Commercial 4.0 International license (CC-BY-NC 4.0) https://creativecommons.org/licenses/by-nc/4.0/

PY - 2023/11/18

Y1 - 2023/11/18

N2 - Background: Lesion resolution is often observed in children with myelin-oligodendrocyte glycoprotein antibody-associated disease (MOGAD), and asymptomatic lesions are less commonly reported in MOGAD than in multiple sclerosis (MS). Objective: We aimed to evaluate brain MRI changes over time in paediatric MOGAD. Methods: Retrospective study in eight UK paediatric neuroscience centres. Acute brain MRI and available follow-up MRIs were reviewed. Predictors for lesion dynamic were evaluated using multivariable regression and Kaplan-Meier survival analyses were used to predict risk of relapse, disability and MOG-Ab status. Results:200 children were included (MOGAD 97; MS 103). At first MRI post-attack, new symptomatic and asymptomatic lesions were seen more often in MS vs MOGAD (52/103 vs 28/97; p=0.002 and 37/103 vs 11/97; p<0.001); 83% of MOGAD patients showed at least one lesion’s resolution at 1st follow‐up scan, and 23% had normal MRI. Only 1 MS patient had single lesionresolution; none had normal MRI. Disappearing lesions in MOGAD were seen in 40% after the 2nd attack, 21% after 3rd attack and none after the 4th attack.New lesions at 1st follow-up scan were associated with increased likelihood of relapse (p=0.02) and persistent MOG-Ab serostatus (p=0.0016) compared to those with no new lesions. Plasma exchange was associated with increased likelihood of lesion resolution (p=0.01). Longer time from symptom onset to steroids was associated with increased likelihood of new lesions; 50% increase at 20 days (p=0.01).Conclusions and Relevance: These striking differences in lesion dynamics between MOGAD and MS suggest greater potential to repair. Early treatment with steroids and plasma exchange is associated with reduced likelihood of new lesions.

AB - Background: Lesion resolution is often observed in children with myelin-oligodendrocyte glycoprotein antibody-associated disease (MOGAD), and asymptomatic lesions are less commonly reported in MOGAD than in multiple sclerosis (MS). Objective: We aimed to evaluate brain MRI changes over time in paediatric MOGAD. Methods: Retrospective study in eight UK paediatric neuroscience centres. Acute brain MRI and available follow-up MRIs were reviewed. Predictors for lesion dynamic were evaluated using multivariable regression and Kaplan-Meier survival analyses were used to predict risk of relapse, disability and MOG-Ab status. Results:200 children were included (MOGAD 97; MS 103). At first MRI post-attack, new symptomatic and asymptomatic lesions were seen more often in MS vs MOGAD (52/103 vs 28/97; p=0.002 and 37/103 vs 11/97; p<0.001); 83% of MOGAD patients showed at least one lesion’s resolution at 1st follow‐up scan, and 23% had normal MRI. Only 1 MS patient had single lesionresolution; none had normal MRI. Disappearing lesions in MOGAD were seen in 40% after the 2nd attack, 21% after 3rd attack and none after the 4th attack.New lesions at 1st follow-up scan were associated with increased likelihood of relapse (p=0.02) and persistent MOG-Ab serostatus (p=0.0016) compared to those with no new lesions. Plasma exchange was associated with increased likelihood of lesion resolution (p=0.01). Longer time from symptom onset to steroids was associated with increased likelihood of new lesions; 50% increase at 20 days (p=0.01).Conclusions and Relevance: These striking differences in lesion dynamics between MOGAD and MS suggest greater potential to repair. Early treatment with steroids and plasma exchange is associated with reduced likelihood of new lesions.

KW - PAEDIATRIC NEUROLOGY

KW - MYELIN

KW - MRI

KW - NEUROIMMUNOLOGY

KW - MULTIPLE SCLEROSIS

UR - https://jnnp.bmj.com/content/early/2023/11/17/jnnp-2023-332542

UR - http://www.scopus.com/inward/record.url?scp=85178054039&partnerID=8YFLogxK

U2 - 10.1136/jnnp-2023-332542

DO - 10.1136/jnnp-2023-332542

M3 - Article

SN - 0022-3050

JO - Journal of Neurology, Neurosurgery and Psychiatry

JF - Journal of Neurology, Neurosurgery and Psychiatry

M1 - jnnp-2023-332542

ER -

Evolution of brain MRI lesions in paediatric myelin-oligodendrocyte glycoprotein antibody-associated disease (MOGAD) and its relevance to disease course

Abstract

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Keywords

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