Incidence of pediatric neuromyelitis optica spectrum disorder and myelin oligodendrocyte glycoprotein antibody-associated disease in Denmark 2008‒2018

A nationwide, population-based cohort study

Magnus S. Boesen*, Poul E.H. Jensen, Alfred P. Born, Melinda Magyari, Anna C. Nilsson, Christina Hoei-Hansen, Morten Blinkenberg, Finn Sellebjerg

*Kontaktforfatter for dette arbejde

Publikation: Bidrag til tidsskriftTidsskriftartikelForskningpeer review

Resumé

Background: The incidence of pediatric neuromyelitis optica spectrum disorder (NMOSD) and myelin oligodendrocyte glycoprotein (MOG) antibody-associated disease have not been reported previously. Our aim was to estimate the incidence of pediatric NMOSD and the occurrence of anti-MOG antibody-associated disease in Denmark during 2008–18, and to evaluate the diagnostic usefulness of antibodies against MOG and aquaporin-4 (AQP4) in children <18 years. Methods: We undertook a nationwide, population-based, multicenter cohort study using data from the Danish National Patient Register, the Danish Multiple Sclerosis Registry, and laboratories providing anti-AQP4 and anti-MOG antibody analyses. Diagnoses were confirmed by review of the medical records, including blinded MRI review in most children with acute disseminated encephalomyelitis (ADEM). Results: In children with acquired demyelinating syndromes, anti-AQP4 antibodies were detected in 4% and anti-MOG antibodies in 18%, including in the two children with ADEM who relapsed. We identified four children with NMOSD, equivalent to an incidence of 0.031/100,000 (95% confidence interval = 0.011‒0.082). In anti-MOG antibody-positive children, 32% relapsed during follow-up. Conclusions: Pediatric NMOSD and MOG antibody-associated disease are rare, but one-third of anti-MOG-positive children relapsed. In pediatric ADEM, only anti-MOG antibody-positive children relapsed, but the overall risk of relapse after pediatric ADEM was low.

OriginalsprogEngelsk
TidsskriftMultiple Sclerosis and Related Disorders
Vol/bind33
Sider (fra-til)162-167
ISSN2211-0348
DOI
StatusUdgivet - 1. aug. 2019

Fingeraftryk

Neuromyelitis Optica
Denmark
Cohort Studies
Pediatrics
Incidence
Population
Rare Diseases
Multicenter Studies
Medical Records
Registries
Confidence Intervals

Citer dette

Boesen, Magnus S. ; Jensen, Poul E.H. ; Born, Alfred P. ; Magyari, Melinda ; Nilsson, Anna C. ; Hoei-Hansen, Christina ; Blinkenberg, Morten ; Sellebjerg, Finn. / Incidence of pediatric neuromyelitis optica spectrum disorder and myelin oligodendrocyte glycoprotein antibody-associated disease in Denmark 2008‒2018 : A nationwide, population-based cohort study. I: Multiple Sclerosis and Related Disorders. 2019 ; Bind 33. s. 162-167.
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title = "Incidence of pediatric neuromyelitis optica spectrum disorder and myelin oligodendrocyte glycoprotein antibody-associated disease in Denmark 2008‒2018: A nationwide, population-based cohort study",
abstract = "Background: The incidence of pediatric neuromyelitis optica spectrum disorder (NMOSD) and myelin oligodendrocyte glycoprotein (MOG) antibody-associated disease have not been reported previously. Our aim was to estimate the incidence of pediatric NMOSD and the occurrence of anti-MOG antibody-associated disease in Denmark during 2008–18, and to evaluate the diagnostic usefulness of antibodies against MOG and aquaporin-4 (AQP4) in children <18 years. Methods: We undertook a nationwide, population-based, multicenter cohort study using data from the Danish National Patient Register, the Danish Multiple Sclerosis Registry, and laboratories providing anti-AQP4 and anti-MOG antibody analyses. Diagnoses were confirmed by review of the medical records, including blinded MRI review in most children with acute disseminated encephalomyelitis (ADEM). Results: In children with acquired demyelinating syndromes, anti-AQP4 antibodies were detected in 4{\%} and anti-MOG antibodies in 18{\%}, including in the two children with ADEM who relapsed. We identified four children with NMOSD, equivalent to an incidence of 0.031/100,000 (95{\%} confidence interval = 0.011‒0.082). In anti-MOG antibody-positive children, 32{\%} relapsed during follow-up. Conclusions: Pediatric NMOSD and MOG antibody-associated disease are rare, but one-third of anti-MOG-positive children relapsed. In pediatric ADEM, only anti-MOG antibody-positive children relapsed, but the overall risk of relapse after pediatric ADEM was low.",
keywords = "ADEM, aquaporin-4, MOG, NMOSD, Pediatric",
author = "Boesen, {Magnus S.} and Jensen, {Poul E.H.} and Born, {Alfred P.} and Melinda Magyari and Nilsson, {Anna C.} and Christina Hoei-Hansen and Morten Blinkenberg and Finn Sellebjerg",
year = "2019",
month = "8",
day = "1",
doi = "10.1016/j.msard.2019.06.002",
language = "English",
volume = "33",
pages = "162--167",
journal = "Multiple Sclerosis and Related Disorders",
issn = "2211-0348",
publisher = "Elsevier",

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Incidence of pediatric neuromyelitis optica spectrum disorder and myelin oligodendrocyte glycoprotein antibody-associated disease in Denmark 2008‒2018 : A nationwide, population-based cohort study. / Boesen, Magnus S.; Jensen, Poul E.H.; Born, Alfred P.; Magyari, Melinda; Nilsson, Anna C.; Hoei-Hansen, Christina; Blinkenberg, Morten; Sellebjerg, Finn.

I: Multiple Sclerosis and Related Disorders, Bind 33, 01.08.2019, s. 162-167.

Publikation: Bidrag til tidsskriftTidsskriftartikelForskningpeer review

TY - JOUR

T1 - Incidence of pediatric neuromyelitis optica spectrum disorder and myelin oligodendrocyte glycoprotein antibody-associated disease in Denmark 2008‒2018

T2 - A nationwide, population-based cohort study

AU - Boesen, Magnus S.

AU - Jensen, Poul E.H.

AU - Born, Alfred P.

AU - Magyari, Melinda

AU - Nilsson, Anna C.

AU - Hoei-Hansen, Christina

AU - Blinkenberg, Morten

AU - Sellebjerg, Finn

PY - 2019/8/1

Y1 - 2019/8/1

N2 - Background: The incidence of pediatric neuromyelitis optica spectrum disorder (NMOSD) and myelin oligodendrocyte glycoprotein (MOG) antibody-associated disease have not been reported previously. Our aim was to estimate the incidence of pediatric NMOSD and the occurrence of anti-MOG antibody-associated disease in Denmark during 2008–18, and to evaluate the diagnostic usefulness of antibodies against MOG and aquaporin-4 (AQP4) in children <18 years. Methods: We undertook a nationwide, population-based, multicenter cohort study using data from the Danish National Patient Register, the Danish Multiple Sclerosis Registry, and laboratories providing anti-AQP4 and anti-MOG antibody analyses. Diagnoses were confirmed by review of the medical records, including blinded MRI review in most children with acute disseminated encephalomyelitis (ADEM). Results: In children with acquired demyelinating syndromes, anti-AQP4 antibodies were detected in 4% and anti-MOG antibodies in 18%, including in the two children with ADEM who relapsed. We identified four children with NMOSD, equivalent to an incidence of 0.031/100,000 (95% confidence interval = 0.011‒0.082). In anti-MOG antibody-positive children, 32% relapsed during follow-up. Conclusions: Pediatric NMOSD and MOG antibody-associated disease are rare, but one-third of anti-MOG-positive children relapsed. In pediatric ADEM, only anti-MOG antibody-positive children relapsed, but the overall risk of relapse after pediatric ADEM was low.

AB - Background: The incidence of pediatric neuromyelitis optica spectrum disorder (NMOSD) and myelin oligodendrocyte glycoprotein (MOG) antibody-associated disease have not been reported previously. Our aim was to estimate the incidence of pediatric NMOSD and the occurrence of anti-MOG antibody-associated disease in Denmark during 2008–18, and to evaluate the diagnostic usefulness of antibodies against MOG and aquaporin-4 (AQP4) in children <18 years. Methods: We undertook a nationwide, population-based, multicenter cohort study using data from the Danish National Patient Register, the Danish Multiple Sclerosis Registry, and laboratories providing anti-AQP4 and anti-MOG antibody analyses. Diagnoses were confirmed by review of the medical records, including blinded MRI review in most children with acute disseminated encephalomyelitis (ADEM). Results: In children with acquired demyelinating syndromes, anti-AQP4 antibodies were detected in 4% and anti-MOG antibodies in 18%, including in the two children with ADEM who relapsed. We identified four children with NMOSD, equivalent to an incidence of 0.031/100,000 (95% confidence interval = 0.011‒0.082). In anti-MOG antibody-positive children, 32% relapsed during follow-up. Conclusions: Pediatric NMOSD and MOG antibody-associated disease are rare, but one-third of anti-MOG-positive children relapsed. In pediatric ADEM, only anti-MOG antibody-positive children relapsed, but the overall risk of relapse after pediatric ADEM was low.

KW - ADEM

KW - aquaporin-4

KW - MOG

KW - NMOSD

KW - Pediatric

U2 - 10.1016/j.msard.2019.06.002

DO - 10.1016/j.msard.2019.06.002

M3 - Journal article

VL - 33

SP - 162

EP - 167

JO - Multiple Sclerosis and Related Disorders

JF - Multiple Sclerosis and Related Disorders

SN - 2211-0348

ER -