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Original research
Evolution of brain MRI lesions in paediatric myelin-oligodendrocyte glycoprotein antibody-associated disease (MOGAD) and its relevance to disease course
  1. Omar Abdel-mannan1,
  2. Dimitrios Champsas1,2,
  3. Carmen Tur1,3,
  4. Vanessa Lee4,
  5. Sharmila Manivannan5,
  6. Haroon Usman6,
  7. Alison Skippen7,
  8. Ishita Desai8,
  9. Manali Chitre9,
  10. Rob Forsyth10,
  11. Rachel Kneen11,12,
  12. Dipak Ram8,
  13. Sithara Ramdas7,
  14. Thomas Rossor4,
  15. Siobhan West8,
  16. Sukhvir Wright13,14,
  17. Jacqueline Palace15,
  18. Evangeline Wassmer13,14,
  19. Cheryl Hemingway16,
  20. Ming J Lim4,
  21. Kshitij Mankad17,
  22. Olga Ciccarelli1,18,
  23. Yael Hacohen1,2
  24. on behalf of the UK-Childhood Neuroinflammatory Disease Network
  1. 1 Department of Neuroinflammation, UCL Institute of Neurology, University College London, London, UK
  2. 2 Department of Neurology, Great Ormond Street Hospital for Children, London, UK
  3. 3 Multiple Sclerosis Centre of Catalonia (Cemcat), Vall d’Hebron Institute of Research, Barcelona, Spain
  4. 4 Children’s Neurosciences, Guy's and St Thomas' NHS Foundation Trust, London, UK
  5. 5 Department of Paediatric Neurology, Addenbrooke’s Hospital, Cambridge, UK
  6. 6 Department of Paediatric Neurology, The Great North Children’s Hospital, Newcastle Upon Tyne, UK
  7. 7 Department of Paediatric Neurology, Oxford Radcliffe Hospitals NHS Trust, Oxford, UK
  8. 8 Department of Neurology, Royal Manchester Children’s Hospital, Manchester, UK
  9. 9 Paediatric Neurology, Addenbrooke's Hospital, Cambridge, UK
  10. 10 Paediatric Neurology, Newcastle Upon Tyne Hospitals NHS Foundation Trust, Newcastle Upon Tyne, UK
  11. 11 Institute of Infection and Global Health, University of Liverpool, Liverpool, UK
  12. 12 Neurology, Alder Hey Children's NHS Foundation Trust, Liverpool, UK
  13. 13 Paediatric Neurology, Birmingham Children's Hospital NHS Foundation Trust, Birmingham, UK
  14. 14 Aston Neuroscience Institute, College of Health and Life Sciences, Aston University, Birmingham, UK
  15. 15 Nuffield Department of Clinical Neurosciences, John Radcliffe Hospital, Oxford, UK
  16. 16 Department of Paediatric Neurology, Great Ormond Street Hospital, London, UK
  17. 17 Neuroradiology, Great Ormond Street Hospital for Children NHS Trust, London, UK
  18. 18 NIHR University College London Hospitals Biomedical Research Centre, London, UK
  1. Correspondence to Dr Yael Hacohen, Department of Neurology, Great Ormond Street Hospital for Children, London, WC1N 3JH, UK; y.hacohen{at}ucl.ac.uk

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 versus MOGAD (52/103 vs 28/97; p=0.002 and 37/103 vs 11/97; p<0.001); 83% of patients with MOGAD showed at least one lesion’s resolution at first follow‐up scan, and 23% had normal MRI. Only 1 patient with MS had single lesion resolution; none had normal MRI. Disappearing lesions in MOGAD were seen in 40% after the second attack, 21% after third attack and none after the fourth attack.

New lesions at first follow-up scan were associated with increased likelihood of relapse (p=0.02) and persistent MOG-Ab serostatus (p=0.0016) compared with 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 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.

  • MRI
  • MULTIPLE SCLEROSIS
  • NEUROIMMUNOLOGY
  • PAEDIATRIC NEUROLOGY
  • MYELIN

Data availability statement

Data are available upon reasonable request. For purposes of replicating procedures and results, any qualified investigator can request anonymised data after ethics clearance and approval by all authors.

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Data availability statement

Data are available upon reasonable request. For purposes of replicating procedures and results, any qualified investigator can request anonymised data after ethics clearance and approval by all authors.

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Footnotes

  • OA-m and DC are joint first authors.

  • X @CarmenTur1

  • Contributors OA-M, DC and YH had full access to all the data in the study and take responsibility for the integrity of the data and the accuracy of the data analysis. YH is responsible for the overall content as the guarantor. Concept and design: YH. Acquisition, analysis, or interpretation of data: all authors. Drafting of the manuscript: OA-M, DC and YH. Critical revision of the manuscript for important intellectual content: JP, OC and YH. Statistical analysis: OA-m, DC, CT and YH. Supervision: YH.

  • Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.

  • Competing interests OA-m receives funding from the Association British Neurologists, MS Society and the Berkeley Foundation’s AW Pidgley Memorial Trust. RF has received grant funding from the NIHR Efficacy and Mechanism Evaluation Programme. CH has received educational and travel grants from Merck Serono and Bayer and Biogen. ML receives research grants from Action Medical Research, the DES society, GOSH charity, National Institute for Health Research, MS Society, and SPARKS charity; receives research support grants from the London Clinical Research Network and Evelina Appeal; has received consultation fees from CSL Behring, Novartis and Octapharma; received travel grants from Merck Serono. OC is a member of independent DSMB for Novartis, gave a teaching talk on McDonald criteria in a Merck local symposium, and contributed to an Advisory Board for Biogen; she is Deputy Editor of Neurology, for which she receives an honorarium.

  • Provenance and peer review Not commissioned; externally peer reviewed.

  • Supplemental material This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.