Long term outcome in non-multiple sclerosis paediatric acquired demyelinating syndromes

Evangeline Wassmer; Charly Billaud; Michael Absoud; Omar Abdel-Mannan; Christina Benetou; Carole Cummins; Katharine Forrest; Christian De Goede; Noha Eltantawi; Helga Hickson; Nahin Hussain; Phil Jardine; John H Livingston; Santosh Mordekar; Sithara Ramdas; Micheal Taylor; K Vijayakumar; Siobhan West; William P Whitehouse; Rachel Kneen; Cheryl Hemingway; Ming Lim; Yael Hacohen; Sukhvir Wright

doi:10.1016/j.ejpn.2024.07.002

Long term outcome in non-multiple sclerosis paediatric acquired demyelinating syndromes

Evangeline Wassmer, Charly Billaud, Michael Absoud, Omar Abdel-Mannan, Christina Benetou, Carole Cummins, Katharine Forrest, Christian De Goede, Noha Eltantawi, Helga Hickson, Nahin Hussain, Phil Jardine, John H Livingston, Santosh Mordekar, Sithara Ramdas, Micheal Taylor, K Vijayakumar, Siobhan West, William P Whitehouse, Rachel KneenCheryl Hemingway, Ming Lim, Yael Hacohen, Sukhvir Wright

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

5 Citations (SciVal)

16 Downloads (Pure)

Abstract

OBJECTIVES: We aimed to study the risks of relapse and long term disability in children with non-MS acquired demyelinating syndromes (ADS).

METHODS: In this prospective, multi-centre study, from the 14 UK pediatric neurology centres, children (<16 years) experiencing a first episode of ADS were recruited from 2010 to 2014. Case report forms were collected prospectively.

RESULTS: A total of 269 children were recruited and followed up for a median of 7.2 years. Median age at onset was 9y (IQR 9.5-14.5, 126 females). At last follow-up, 46 (18 %) had MS, 4 AQP4-Ab NMOSD and 206 (80 %) had other ADS, of which 27 (13 %) relapsed. Relapsing MOGAD was the diagnosis in 12/27, 6 were seronegative and 9 did not have antibodies tested. Frequency of relapse differed according to first presentation in non-MS ADS, being least likely in transverse myelitis (p = 0.025). In the non-MS group, MOG-Ab was predictive of relapse (HR = 8.42; p < 0.001) occurring 8 times as often decreasing over time. Long-term difficulties did not differ between children with monophasic vs relapsing diseases.

CONCLUSION: The risk of relapse in non-MS ADS depends on initial diagnosis, and MOG-Ab positivity. Long-term difficulties are observed regardless of relapses and are determined by presenting phenotype.

Original language	English
Pages (from-to)	52-58
Number of pages	7
Journal	European Journal of Paediatric Neurology
Volume	52
Early online date	6 Jul 2024
DOIs	https://doi.org/10.1016/j.ejpn.2024.07.002
Publication status	Published - Sept 2024

Bibliographical note

Copyright © 2024. Published by Elsevier Ltd on behalf of European Paediatric Neurology Society. This work is licensed under a Creative Commons Attribution 4.0 International License (https://creativecommons.org/licenses/by/4.0/).

Funding

This study is funded by a grant from the UK Multiple Sclerosis Society (893/08) and Action Medical Research (SP4472). Also, funding was received from Epilepsy Research UK, Encephalitis Society and BCH Charity Research Fund. The protocol was also developed and approved by the UK Children’s Neurological Research Campaign. The study was supported by the Medicines for Children Research Network and the Birmingham Children’s Hospital Wellcome Trust Clinical Research Facility

Keywords

AQP4-Ab NMOSD
Acquired demyelinating syndromes
Long term outcomes
MOGAD
Multiple sclerosis

Access to Document

10.1016/j.ejpn.2024.07.002Licence: CC BY 4.0

E Wassmer et al_Long term outcome in non-multiple sclerosis paediatric acquired demyelinating syndromes
Copyright © 2024. Published by Elsevier Ltd on behalf of European Paediatric Neurology Society. This work is licensed under a Creative Commons Attribution 4.0 International License (https://creativecommons.org/licenses/by/4.0/).
Final published version, 1.08 MBLicence: CC BY 4.0

https://discovery.ucl.ac.uk/id/eprint/10194412/

Cite this

Wassmer, E., Billaud, C., Absoud, M., Abdel-Mannan, O., Benetou, C., Cummins, C., Forrest, K., De Goede, C., Eltantawi, N., Hickson, H., Hussain, N., Jardine, P., Livingston, J. H., Mordekar, S., Ramdas, S., Taylor, M., Vijayakumar, K., West, S., Whitehouse, W. P., ... Wright, S. (2024). Long term outcome in non-multiple sclerosis paediatric acquired demyelinating syndromes. European Journal of Paediatric Neurology, 52, 52-58. https://doi.org/10.1016/j.ejpn.2024.07.002

@article{d4002dcdb80f4a9aad0754527dd22e3d,

title = "Long term outcome in non-multiple sclerosis paediatric acquired demyelinating syndromes",

abstract = "OBJECTIVES: We aimed to study the risks of relapse and long term disability in children with non-MS acquired demyelinating syndromes (ADS).METHODS: In this prospective, multi-centre study, from the 14 UK pediatric neurology centres, children (<16 years) experiencing a first episode of ADS were recruited from 2010 to 2014. Case report forms were collected prospectively.RESULTS: A total of 269 children were recruited and followed up for a median of 7.2 years. Median age at onset was 9y (IQR 9.5-14.5, 126 females). At last follow-up, 46 (18 %) had MS, 4 AQP4-Ab NMOSD and 206 (80 %) had other ADS, of which 27 (13 %) relapsed. Relapsing MOGAD was the diagnosis in 12/27, 6 were seronegative and 9 did not have antibodies tested. Frequency of relapse differed according to first presentation in non-MS ADS, being least likely in transverse myelitis (p = 0.025). In the non-MS group, MOG-Ab was predictive of relapse (HR = 8.42; p < 0.001) occurring 8 times as often decreasing over time. Long-term difficulties did not differ between children with monophasic vs relapsing diseases.CONCLUSION: The risk of relapse in non-MS ADS depends on initial diagnosis, and MOG-Ab positivity. Long-term difficulties are observed regardless of relapses and are determined by presenting phenotype.",

keywords = "AQP4-Ab NMOSD, Acquired demyelinating syndromes, Long term outcomes, MOGAD, Multiple sclerosis",

author = "Evangeline Wassmer and Charly Billaud and Michael Absoud and Omar Abdel-Mannan and Christina Benetou and Carole Cummins and Katharine Forrest and {De Goede}, Christian and Noha Eltantawi and Helga Hickson and Nahin Hussain and Phil Jardine and Livingston, {John H} and Santosh Mordekar and Sithara Ramdas and Micheal Taylor and K Vijayakumar and Siobhan West and Whitehouse, {William P} and Rachel Kneen and Cheryl Hemingway and Ming Lim and Yael Hacohen and Sukhvir Wright",

note = "Copyright {\textcopyright} 2024. Published by Elsevier Ltd on behalf of European Paediatric Neurology Society. This work is licensed under a Creative Commons Attribution 4.0 International License (https://creativecommons.org/licenses/by/4.0/).",

year = "2024",

month = sep,

doi = "10.1016/j.ejpn.2024.07.002",

language = "English",

volume = "52",

pages = "52--58",

journal = "European Journal of Paediatric Neurology",

issn = "1090-3798",

publisher = "Elsevier",

}

Wassmer, E, Billaud, C, Absoud, M, Abdel-Mannan, O, Benetou, C, Cummins, C, Forrest, K, De Goede, C, Eltantawi, N, Hickson, H, Hussain, N, Jardine, P, Livingston, JH, Mordekar, S, Ramdas, S, Taylor, M, Vijayakumar, K, West, S, Whitehouse, WP, Kneen, R, Hemingway, C, Lim, M, Hacohen, Y & Wright, S 2024, 'Long term outcome in non-multiple sclerosis paediatric acquired demyelinating syndromes', European Journal of Paediatric Neurology, vol. 52, pp. 52-58. https://doi.org/10.1016/j.ejpn.2024.07.002

TY - JOUR

T1 - Long term outcome in non-multiple sclerosis paediatric acquired demyelinating syndromes

AU - Wassmer, Evangeline

AU - Billaud, Charly

AU - Absoud, Michael

AU - Abdel-Mannan, Omar

AU - Benetou, Christina

AU - Cummins, Carole

AU - Forrest, Katharine

AU - De Goede, Christian

AU - Eltantawi, Noha

AU - Hickson, Helga

AU - Hussain, Nahin

AU - Jardine, Phil

AU - Livingston, John H

AU - Mordekar, Santosh

AU - Ramdas, Sithara

AU - Taylor, Micheal

AU - Vijayakumar, K

AU - West, Siobhan

AU - Whitehouse, William P

AU - Kneen, Rachel

AU - Hemingway, Cheryl

AU - Lim, Ming

AU - Hacohen, Yael

AU - Wright, Sukhvir

N1 - Copyright © 2024. Published by Elsevier Ltd on behalf of European Paediatric Neurology Society. This work is licensed under a Creative Commons Attribution 4.0 International License (https://creativecommons.org/licenses/by/4.0/).

PY - 2024/9

Y1 - 2024/9

N2 - OBJECTIVES: We aimed to study the risks of relapse and long term disability in children with non-MS acquired demyelinating syndromes (ADS).METHODS: In this prospective, multi-centre study, from the 14 UK pediatric neurology centres, children (<16 years) experiencing a first episode of ADS were recruited from 2010 to 2014. Case report forms were collected prospectively.RESULTS: A total of 269 children were recruited and followed up for a median of 7.2 years. Median age at onset was 9y (IQR 9.5-14.5, 126 females). At last follow-up, 46 (18 %) had MS, 4 AQP4-Ab NMOSD and 206 (80 %) had other ADS, of which 27 (13 %) relapsed. Relapsing MOGAD was the diagnosis in 12/27, 6 were seronegative and 9 did not have antibodies tested. Frequency of relapse differed according to first presentation in non-MS ADS, being least likely in transverse myelitis (p = 0.025). In the non-MS group, MOG-Ab was predictive of relapse (HR = 8.42; p < 0.001) occurring 8 times as often decreasing over time. Long-term difficulties did not differ between children with monophasic vs relapsing diseases.CONCLUSION: The risk of relapse in non-MS ADS depends on initial diagnosis, and MOG-Ab positivity. Long-term difficulties are observed regardless of relapses and are determined by presenting phenotype.

AB - OBJECTIVES: We aimed to study the risks of relapse and long term disability in children with non-MS acquired demyelinating syndromes (ADS).METHODS: In this prospective, multi-centre study, from the 14 UK pediatric neurology centres, children (<16 years) experiencing a first episode of ADS were recruited from 2010 to 2014. Case report forms were collected prospectively.RESULTS: A total of 269 children were recruited and followed up for a median of 7.2 years. Median age at onset was 9y (IQR 9.5-14.5, 126 females). At last follow-up, 46 (18 %) had MS, 4 AQP4-Ab NMOSD and 206 (80 %) had other ADS, of which 27 (13 %) relapsed. Relapsing MOGAD was the diagnosis in 12/27, 6 were seronegative and 9 did not have antibodies tested. Frequency of relapse differed according to first presentation in non-MS ADS, being least likely in transverse myelitis (p = 0.025). In the non-MS group, MOG-Ab was predictive of relapse (HR = 8.42; p < 0.001) occurring 8 times as often decreasing over time. Long-term difficulties did not differ between children with monophasic vs relapsing diseases.CONCLUSION: The risk of relapse in non-MS ADS depends on initial diagnosis, and MOG-Ab positivity. Long-term difficulties are observed regardless of relapses and are determined by presenting phenotype.

KW - AQP4-Ab NMOSD

KW - Acquired demyelinating syndromes

KW - Long term outcomes

KW - MOGAD

KW - Multiple sclerosis

UR - https://linkinghub.elsevier.com/retrieve/pii/S1090379824000990

UR - http://www.scopus.com/inward/record.url?scp=85198569708&partnerID=8YFLogxK

U2 - 10.1016/j.ejpn.2024.07.002

DO - 10.1016/j.ejpn.2024.07.002

M3 - Article

C2 - 39025036

SN - 1090-3798

VL - 52

SP - 52

EP - 58

JO - European Journal of Paediatric Neurology

JF - European Journal of Paediatric Neurology

ER -

Long term outcome in non-multiple sclerosis paediatric acquired demyelinating syndromes

Abstract

Bibliographical note

Funding

Keywords

Access to Document

Other files and links

Fingerprint

Cite this