@article{ca1ec305dbec404d814092efb4b2ef05,
title = "Incidence of paediatric multiple sclerosis and other acquired demyelinating syndromes: 10-year follow-up surveillance study",
abstract = "Aim: To describe a 10-year follow-up of children (<16y) with acquired demyelinating syndromes (ADS) from a UK-wide prospective surveillance study. Method: Diagnoses were retrieved from the patients' records via the patients' paediatric or adult neurologist using a questionnaire. Demyelinating phenotypes at follow-up were classified by an expert review panel. Results: Twenty-four out of 125 (19.2%) children (64 males, 61 females; median age 10y, range 1y 4mo–15y 11mo), identified in the original study, were diagnosed with multiple sclerosis (incidence of 2.04/million children/year); 23 of 24 fulfilled 2017 McDonald criteria at onset. Aquaporin-4-antibody neuromyelitis optica spectrum disorders were diagnosed in three (2.4%, 0.26/million children/year), and relapsing myelin oligodendrocyte glycoprotein antibody-associated disease in five (4%, 0.43/million children/year). Three out of 125 seronegative patients relapsed and 85 of 125 (68%) remained monophasic over 10 years. Five of 125 patients (4%) originally diagnosed with ADS were reclassified during follow-up: three children diagnosed initially with acute disseminated encephalomyelitis were subsequently diagnosed with acute necrotising encephalopathy (RAN-binding protein 2 mutation), primary haemophagocytic lymphohistiocytosis (Munc 13-4 gene inversion), and anti-N-methyl-d-aspartate receptor encephalitis. One child initially diagnosed with optic neuritis was later diagnosed with vitamin B12 deficiency, and one presenting with transverse myelitis was subsequently diagnosed with Sj{\"o}gren syndrome. Interpretation: The majority of ADS presentations in children are monophasic, even at 10-year follow-up. Given the implications for treatment strategies, multiple sclerosis and central nervous system autoantibody mimics warrant extensive investigation.",
keywords = "Autoantibodies, Child, Female, Follow-Up Studies, Humans, Incidence, Male, Multiple Sclerosis/diagnosis, Myelin-Oligodendrocyte Glycoprotein, Neoplasm Recurrence, Local, Prospective Studies, Syndrome",
author = "{UK Childhood Neuroinflammatory Disorders Network} and Omar Abdel-Mannan and Michael Absoud and Christina Benetou and Helga Hickson and Cheryl Hemingway and Ming Lim and Sukhvir Wright and Yael Hacohen and Evangeline Wassmer",
note = "Funding Information: We are grateful to Olga Ciccarelli for her thorough reviews and comments on the manuscript. The British Paediatric Surveillance Unit's and British Ophthalmological Surveillance Unit's roles in facilitation of data collection is acknowledged. Cheryl Hemingway has received educational and travel grants from Merck Serono and Bayer and Biogen. Ming Lim receives research grants from Action Medical Research, the Dancing Eyes Syndrome society, Great Ormond Street Hospital charity, National Institute for Health Research, MS Society, and the 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; has received travel grants from Merck Serono. Evangeline Wassmer receives research grants from Action Medical Research and the MS Society; has received travel grants from UCB, Shire and Biogen Idec; educational grants to organize meetings from Merck Serono, Novartis, Bayer and Biogen Idec; speaker's fees from Merck Serono; and consultancy fees from Genzyme. Sukhvir Wright received grants and funding from Epilepsy Research UK, The Wellcome Trust, The Encephalitis Society, Birmingham Women's and Children's Hospital Research Funds. The original study was funded by the Multiple Sclerosis Society UK (grant number 893 of 08) and Action Medical Research (grant number SP4472). The study also received support from the Wellcome Trust (grant number GN79832) which funded the Birmingham Children's Hospital research facility. Omar Abdel‐Mannan is funded by a Guarantors of Brain Clinical Entry Fellowship. Yael Hacohen is funded by the MS Society. Cheryl Hemingway is funded by an MRC Clinical Academic Research Partnership grant. Sukvir Wright is funded by a Wellcome Trust Clinical Research Career Development Fellowship (216613/Z/19/Z). Publisher Copyright: {\textcopyright} 2021 Mac Keith Press",
year = "2022",
month = apr,
doi = "10.1111/dmcn.15098",
language = "English",
volume = "64",
pages = "502--508",
journal = "Developmental Medicine and Child Neurology",
issn = "0012-1622",
publisher = "Wiley-Blackwell",
number = "4",
}