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

C. L. de Mol; Y. Y. M. Wong; E. D. van Pelt; I. A. Ketelslegers; D. P. Bakker; M. Boon; K. P. J. Braun; K. G. J. van Dijk; M. J. Eikelenboom; M. Engelen; K. Geleijns; C. A. Haaxma; J. M. F. Niermeijer; E. H. Niks; E. A. J. Peeters; C. M. P. C. D. Peeters-Scholte; B. T. Poll-The; R. P. Portier; J. F. de Rijk-van Andel; J. P. A. Samijn; H. M. Schippers; I. N. Snoeck; H. Stroink; R. J. Vermeulen; A. Verrips; F. Visscher; J. S. H. Vles; M. A. A. P. Willemsen; C. E. Catsman-Berrevoets; R. Q. Hintzen; R. F. Neuteboom

doi:10.1007/s00415-018-8835-6

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

C. L. de Mol, Y. Y. M. Wong, E. D. van Pelt, I. A. Ketelslegers, D. P. Bakker, M. Boon, K. P. J. Braun, K. G. J. van Dijk, M. J. Eikelenboom, M. Engelen, K. Geleijns, C. A. Haaxma, J. M. F. Niermeijer, E. H. Niks, E. A. J. Peeters, C. M. P. C. D. Peeters-Scholte, B. T. Poll-The, R. P. Portier, J. F. de Rijk-van Andel, J. P. A. SamijnH. M. Schippers, I. N. Snoeck, H. Stroink, R. J. Vermeulen, A. Verrips, F. Visscher, J. S. H. Vles, M. A. A. P. Willemsen, C. E. Catsman-Berrevoets, R. Q. Hintzen, R. F. Neuteboom^*

^*Corresponding author for this work

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

Abstract

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. 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. 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 ae< 5.5 was reached in 3/89 MS patients (3%). 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.

Original language	English
Pages (from-to)	1310-1319
Number of pages	10
Journal	Journal of Neurology
Volume	265
Issue number	6
DOIs	https://doi.org/10.1007/s00415-018-8835-6
Publication status	Published - 1 Jun 2018

Keywords

Children
Multiple sclerosis
Acquired demyelinating syndromes
Epidemiology
Outcome
ACUTE DISSEMINATED ENCEPHALOMYELITIS
PEDIATRIC MULTIPLE-SCLEROSIS
CHILDHOOD-ONSET
FOLLOW-UP
CNS
ANTIBODIES
FEATURES
DEFINITIONS
ADOLESCENTS
IMPAIRMENT
Prospective Studies
Follow-Up Studies
Humans
Child, Preschool
Male
Central Nervous System Diseases/epidemiology
Demyelinating Diseases/epidemiology
Incidence
Female
Netherlands/epidemiology
Child
Adolescent

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de Mol, C. L., Wong, Y. Y. M., van Pelt, E. D., Ketelslegers, I. A., Bakker, D. P., Boon, M., Braun, K. P. J., van Dijk, K. G. J., Eikelenboom, M. J., Engelen, M., Geleijns, K., Haaxma, C. A., Niermeijer, J. M. F., Niks, E. H., Peeters, E. A. J., Peeters-Scholte, C. M. P. C. D., Poll-The, B. T., Portier, R. P., de Rijk-van Andel, J. F., ... Neuteboom, R. F. (2018). Incidence and outcome of acquired demyelinating syndromes in Dutch children: update of a nationwide and prospective study. Journal of Neurology, 265(6), 1310-1319. https://doi.org/10.1007/s00415-018-8835-6

@article{bac7e8f5a0ad4d0db3405ff7cfd88b32,

title = "Incidence and outcome of acquired demyelinating syndromes in Dutch children: update of a nationwide and prospective study",

abstract = "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. 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. 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 ae< 5.5 was reached in 3/89 MS patients (3%). 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.",

keywords = "Children, Multiple sclerosis, Acquired demyelinating syndromes, Epidemiology, Outcome, ACUTE DISSEMINATED ENCEPHALOMYELITIS, PEDIATRIC MULTIPLE-SCLEROSIS, CHILDHOOD-ONSET, FOLLOW-UP, CNS, ANTIBODIES, FEATURES, DEFINITIONS, ADOLESCENTS, IMPAIRMENT, Prospective Studies, Follow-Up Studies, Humans, Child, Preschool, Male, Central Nervous System Diseases/epidemiology, Demyelinating Diseases/epidemiology, Incidence, Female, Netherlands/epidemiology, Child, Adolescent",

author = "{de Mol}, {C. L.} and Wong, {Y. Y. M.} and {van Pelt}, {E. D.} and Ketelslegers, {I. A.} and Bakker, {D. P.} and M. Boon and Braun, {K. P. J.} and {van Dijk}, {K. G. J.} and Eikelenboom, {M. J.} and M. Engelen and K. Geleijns and Haaxma, {C. A.} and Niermeijer, {J. M. F.} and Niks, {E. H.} and Peeters, {E. A. J.} and Peeters-Scholte, {C. M. P. C. D.} and Poll-The, {B. T.} and Portier, {R. P.} and {de Rijk-van Andel}, {J. F.} and Samijn, {J. P. A.} and Schippers, {H. M.} and Snoeck, {I. N.} and H. Stroink and Vermeulen, {R. J.} and A. Verrips and F. Visscher and Vles, {J. S. H.} and Willemsen, {M. A. A. P.} and Catsman-Berrevoets, {C. E.} and Hintzen, {R. Q.} and Neuteboom, {R. F.}",

year = "2018",

month = jun,

day = "1",

doi = "10.1007/s00415-018-8835-6",

language = "English",

volume = "265",

pages = "1310--1319",

journal = "Journal of Neurology",

issn = "0340-5354",

publisher = "Springer",

number = "6",

}

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 & Neuteboom, RF 2018, 'Incidence and outcome of acquired demyelinating syndromes in Dutch children: update of a nationwide and prospective study', Journal of Neurology, vol. 265, no. 6, pp. 1310-1319. https://doi.org/10.1007/s00415-018-8835-6

TY - JOUR

T1 - Incidence and outcome of acquired demyelinating syndromes in Dutch children

T2 - update of a nationwide and prospective study

AU - de Mol, C. L.

AU - Wong, Y. Y. M.

AU - van Pelt, E. D.

AU - Ketelslegers, I. A.

AU - Bakker, D. P.

AU - Boon, M.

AU - Braun, K. P. J.

AU - van Dijk, K. G. J.

AU - Eikelenboom, M. J.

AU - Engelen, M.

AU - Geleijns, K.

AU - Haaxma, C. A.

AU - Niermeijer, J. M. F.

AU - Niks, E. H.

AU - Peeters, E. A. J.

AU - Peeters-Scholte, C. M. P. C. D.

AU - Poll-The, B. T.

AU - Portier, R. P.

AU - de Rijk-van Andel, J. F.

AU - Samijn, J. P. A.

AU - Schippers, H. M.

AU - Snoeck, I. N.

AU - Stroink, H.

AU - Vermeulen, R. J.

AU - Verrips, A.

AU - Visscher, F.

AU - Vles, J. S. H.

AU - Willemsen, M. A. A. P.

AU - Catsman-Berrevoets, C. E.

AU - Hintzen, R. Q.

AU - Neuteboom, R. F.

PY - 2018/6/1

Y1 - 2018/6/1

N2 - 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. 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. 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 ae< 5.5 was reached in 3/89 MS patients (3%). 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.

AB - 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. 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. 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 ae< 5.5 was reached in 3/89 MS patients (3%). 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.

KW - Children

KW - Multiple sclerosis

KW - Acquired demyelinating syndromes

KW - Epidemiology

KW - Outcome

KW - ACUTE DISSEMINATED ENCEPHALOMYELITIS

KW - PEDIATRIC MULTIPLE-SCLEROSIS

KW - CHILDHOOD-ONSET

KW - FOLLOW-UP

KW - CNS

KW - ANTIBODIES

KW - FEATURES

KW - DEFINITIONS

KW - ADOLESCENTS

KW - IMPAIRMENT

KW - Prospective Studies

KW - Follow-Up Studies

KW - Humans

KW - Child, Preschool

KW - Male

KW - Central Nervous System Diseases/epidemiology

KW - Demyelinating Diseases/epidemiology

KW - Incidence

KW - Female

KW - Netherlands/epidemiology

KW - Child

KW - Adolescent

U2 - 10.1007/s00415-018-8835-6

DO - 10.1007/s00415-018-8835-6

M3 - Article

C2 - 29569176

SN - 0340-5354

VL - 265

SP - 1310

EP - 1319

JO - Journal of Neurology

JF - Journal of Neurology

IS - 6

ER -

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

Abstract

Keywords

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