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1. Incidence and outcome of acquired demyelinating syndromes in Dutch children: update of a nationwide and prospective study

Author

Mol, Louk, Wong, Yu Yi, van Pelt - T Gravesteijn, Elles, Ketelslegers, IA, Bakker, DP, Boon, M, Braun, KPJ, van Dijk, KGJ, Eikelenboom, MJ, Engelen, MP, Geleijns, K, Haaxma, C A, Niermeijer, JMF, Niks, EH, Peeters, EAJ, Peeters-Scholte, C, Poll-The, BT, Portier, RP, De Rijk-Van Andel, JF, Samijn, JPA, Schippers, HM, Snoeck, IN, Stroink, H, Vermeulen, RJ, Verrips, A, Visscher, F, Vles, JSH, Willemsen, M, Catsman - Berrevoets, Coriene, Hintzen, Rogier, Neuteboom, Rinze, Mol, Louk, Wong, Yu Yi, van Pelt - T Gravesteijn, Elles, Ketelslegers, IA, Bakker, DP, Boon, M, Braun, KPJ, van Dijk, KGJ, Eikelenboom, MJ, Engelen, MP, Geleijns, K, Haaxma, C A, Niermeijer, JMF, Niks, EH, Peeters, EAJ, Peeters-Scholte, C, Poll-The, BT, Portier, RP, De Rijk-Van Andel, JF, Samijn, JPA, Schippers, HM, Snoeck, IN, Stroink, H, Vermeulen, RJ, Verrips, A, Visscher, F, Vles, JSH, Willemsen, M, Catsman - Berrevoets, Coriene, Hintzen, Rogier, and Neuteboom, Rinze

Published
2018

2. Incidence of acquired demyelinating syndromes of the CNS in Dutch children: a nationwide study

Author

Ketelslegers, Immy, Catsman - Berrevoets, Coriene, Neuteboom, Rinze, Boon, M, van Dijk, KGJ, Eikelenboom, MJ, Gooskens, RHJM, Niks, EH (Erik), Overweg-Plandsoen, WCG, Peeters, EAJ, Peeters-Scholte, CMPCD, Poll-The, BT, De Rijk-Van Andel, JF, Samijn, JPA, Snoeck, IN, Stroink, H (Hans), Vermeulen, RJ, Verrips, A, Vles, JSH, Willemsen, MAAP, Pereira, RR, Hintzen, Rogier, Ketelslegers, Immy, Catsman - Berrevoets, Coriene, Neuteboom, Rinze, Boon, M, van Dijk, KGJ, Eikelenboom, MJ, Gooskens, RHJM, Niks, EH (Erik), Overweg-Plandsoen, WCG, Peeters, EAJ, Peeters-Scholte, CMPCD, Poll-The, BT, De Rijk-Van Andel, JF, Samijn, JPA, Snoeck, IN, Stroink, H (Hans), Vermeulen, RJ, Verrips, A, Vles, JSH, Willemsen, MAAP, Pereira, RR, and Hintzen, Rogier

Abstract

Acquired demyelinating syndromes (ADS) can be a first presentation of multiple sclerosis (MS) in children. The incidence of these disorders in Europe is currently unknown. Children (< 18 years old) living in the Netherlands who presented with ADS were included from January 1, 2007 to December 31, 2010 by the Dutch pediatric MS study group and the Dutch surveillance of rare pediatric disorders. Demographic and clinical data were collected. Eighty-six patients were identified over 4 years, resulting in an incidence of 0.66/1,00,000 per year. Most patients presented with polyfocal ADS without encephalopathy (30%), followed by polyfocal ADS with encephalopathy (24%), optic neuritis (ON, 22%), monofocal ADS (16%), transverse myelitis (3%), and neuromyelitis optica (3%). Patients with polyfocal ADS with encephalopathy were younger (median 3.9 years) than patients with ON (median 14.6 years, p < 0.001) or monofocal ADS (median 16.0 years, p < 0.001). Patients with polyfocal ADS without encephalopathy (median 9.2 years) were also younger than monofocal ADS patients (median 16.0 years, p < 0.001). There was a slight female preponderance in all groups except the ON group, and a relatively large number of ADS patients (29%) reported a non-European ancestry. Familial autoimmune diseases were reported in 23%, more often in patients with relapsing disease than monophasic disease (46 vs. 15%, p = 0.002) and occurring most often in the maternal family (84%, p < 0.001). During the study period, 23% of patients were subsequently diagnosed with MS. The annual incidence of ADS in the Netherlands is 0.66/1,00,000 children/year. A polyfocal disease onset of ADS was most common.

Published
2012

3. Incidence and outcome of acquired demyelinating syndromes in Dutch children: update of a nationwide and prospective study.

Author

de Mol CL, Wong YYM, van Pelt ED, Ketelslegers IA, Bakker DP, Boon M, Braun KPJ, van Dijk KGJ, Eikelenboom MJ, Engelen M, Geleijns K, Haaxma CA, Niermeijer JMF, Niks EH, Peeters EAJ, Peeters-Scholte CMPCD, Poll-The BT, Portier RP, de Rijk-van Andel JF, Samijn JPA, Schippers HM, Snoeck IN, Stroink H, Vermeulen RJ, Verrips A, Visscher F, Vles JSH, Willemsen MAAP, Catsman-Berrevoets CE, Hintzen RQ, and Neuteboom RF

Subjects
Adolescent, Central Nervous System Diseases therapy, Child, Child, Preschool, Demyelinating Diseases therapy, Female, Follow-Up Studies, Humans, Incidence, Male, Netherlands epidemiology, Prospective Studies, Central Nervous System Diseases epidemiology, Demyelinating Diseases epidemiology
Abstract

Introduction: Acquired demyelinating syndromes (ADS) are immune-mediated demyelinating disorders of the central nervous system in children. A nationwide, multicentre and prospective cohort study was initiated in the Netherlands in 2006, with a reported ADS incidence of 0.66/100,000 per year and MS incidence of 0.15/100,000 per year in the period between 2007 and 2010. In this study, we provide an update on the incidence and the long-term follow-up of ADS in the Netherlands., Methods: Children < 18 years with a first attack of demyelination were included consecutively from January 2006 to December 2016. Diagnoses were based on the International Paediatric MS study group consensus criteria. Outcome data were collected by neurological and neuropsychological assessments, and telephone call assessments., Results: Between 2011 and 2016, 55/165 of the ADS patients were diagnosed with MS (33%). This resulted in an increased ADS and MS incidence of 0.80/100,000 per year and 0.26/100,000 per year, respectively. Since 2006 a total of 243 ADS patients have been included. During follow-up (median 55 months, IQR 28-84), 137 patients were diagnosed with monophasic disease (56%), 89 with MS (37%) and 17 with multiphasic disease other than MS (7%). At least one form of residual deficit including cognitive impairment was observed in 69% of all ADS patients, even in monophasic ADS. An Expanded Disability Status Scale score of ≥ 5.5 was reached in 3/89 MS patients (3%)., Conclusion: The reported incidence of ADS in Dutch children has increased since 2010. Residual deficits are common in this group, even in monophasic patients. Therefore, long-term follow-up in ADS patients is warranted.

Published
2018
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