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 & 11 others 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

Research output: Contribution to journalArticleAcademicpeer-review

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.
Original languageEnglish
Pages (from-to)1310-1319
JournalJournal of Neurology
Volume265
Issue number6
DOIs
Publication statusPublished - 2018

Cite this

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. ; Samijn, J. P. A. ; Schippers, H. M. ; Snoeck, I. N. ; Stroink, H. ; Vermeulen, R. J. ; Verrips, A. ; Visscher, F. ; Vles, J. S. H. ; Willemsen, M. A. A. P. ; Catsman-Berrevoets, C. E. ; Hintzen, R. Q. ; Neuteboom, R. F. / Incidence and outcome of acquired demyelinating syndromes in Dutch children: update of a nationwide and prospective study. In: Journal of Neurology. 2018 ; Vol. 265, No. 6. pp. 1310-1319.
@article{04a2404f7cb74df7b4a21534ce0cab31,
title = "Incidence and outcome of acquired demyelinating syndromes in Dutch children: update of a nationwide and prospective study",
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.",
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",
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volume = "265",
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journal = "Journal of Neurology",
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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

Incidence and outcome of acquired demyelinating syndromes in Dutch children: update of a nationwide and prospective study. / 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.; Samijn, J. P. A.; Schippers, H. M.; Snoeck, I. N.; Stroink, H.; Vermeulen, R. J.; Verrips, A.; Visscher, F.; Vles, J. S. H.; Willemsen, M. A. A. P.; Catsman-Berrevoets, C. E.; Hintzen, R. Q.; Neuteboom, R. F.

In: Journal of Neurology, Vol. 265, No. 6, 2018, p. 1310-1319.

Research output: Contribution to journalArticleAcademicpeer-review

TY - JOUR

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

Y1 - 2018

N2 - 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.

AB - 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.

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UR - https://www.ncbi.nlm.nih.gov/pubmed/29569176

U2 - 10.1007/s00415-018-8835-6

DO - 10.1007/s00415-018-8835-6

M3 - Article

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SP - 1310

EP - 1319

JO - Journal of Neurology

JF - Journal of Neurology

SN - 0340-5354

IS - 6

ER -