Implications of the International Paediatric Multiple Sclerosis Study Group consensus criteria for paediatric acute disseminated encephalomyelitis: a nationwide validation study
Aim The International Paediatric Multiple Sclerosis Study Group (IPMSSG) has proposed criteria for acute disseminated encephalomyelitis (ADEM) not evaluated in clinical practice. Our objective was to assess epidemiological implications of the IPMSSG criteria for ADEM in a cohort study using prospect...
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| Veröffentlicht in: | Developmental medicine and child neurology 2018-11, Vol.60 (11), p.1123-1131 |
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| creator | Boesen, Magnus S Blinkenberg, Morten Koch‐Henriksen, Nils Thygesen, Lau C Uldall, Peter V Magyari, Melinda Born, Alfred P |
| description | Aim
The International Paediatric Multiple Sclerosis Study Group (IPMSSG) has proposed criteria for acute disseminated encephalomyelitis (ADEM) not evaluated in clinical practice. Our objective was to assess epidemiological implications of the IPMSSG criteria for ADEM in a cohort study using prospectively collected data.
Method
We identified all diagnosed cases of ADEM in Denmark between 2008 and 2015 from the Danish National Patient Register by International Classification of Diseases 10 codes assigned to acute demyelinating episodes, and we reviewed all medical records to validate ADEM.
Results
We found 52 children up to the age of 18 years with a verified clinical diagnosis of ADEM (incidence rate 0.54/100 000 person‐years; all had abnormal brain magnetic resonance imaging). Only 18 (35%) fulfilled the IPMSSG criteria regarding encephalopathy and polyfocal neurological deficits. Among all 52 children with ADEM, 33 per cent had clinical sequelae after a median follow‐up of 4 years 6 months (range: 10mo–8y 3mo). Surprisingly, none progressed to multiphasic ADEM or multiple sclerosis, but median age at end of follow‐up was only 10 years 9 months (range: 2y–24y 3mo).
Interpretation
Among 52 children with ADEM, none converted to multiphasic ADEM or multiple sclerosis (median follow‐up: 4y 6mo; range: 10mo–8y 3mo). Applying the IPMSSG criteria to all children with a diagnosis of ADEM leaves 65 per cent of the cases without a diagnosis and lowers the incidence rate of paediatric ADEM.
What this paper adds
The incidence of paediatric acute disseminated encephalomyelitis (ADEM) was 0.54 per 100 000 person‐years in children younger than 18 years.
Only 35 per cent of children with ADEM fulfilled the International Paediatric Study Group consensus criteria.
ADEM in clinical practice was primarily based on magnetic resonance imaging findings.
Paediatric neurologists diagnosed ADEM in the absence of encephalopathy.
None of the children with ADEM progressed to multiple sclerosis/multiphasic ADEM during follow‐up.
Resumen
Implicancias en los criterios del consenso del grupo de estudio internacional de esclerosis múltiple pediátrico para la encefalitis aguda diseminada pediátrica: un estudio de validación nacional
Objetivos
El grupo de estudios internacional de esclerosis múltiple pediátrico (IPMSSG) ha propuesto criterios para la encefalitis aguda diseminada (ADEM) no evaluada en la práctica clínica. Nuestro objetivo es evaluar las implicancias epidemiológicas de los cri |
| doi_str_mv | 10.1111/dmcn.13798 |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_miscellaneous_2037054246</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><sourcerecordid>2037054246</sourcerecordid><originalsourceid>FETCH-LOGICAL-c3658-53e2d7c381d479607ff0b2e35dcce5c92f3f45ca6be5c8f0068f1dfa84a7de2f3</originalsourceid><addsrcrecordid>eNp9kcuOFCEUhonROO3oxgcwZ2lMagSKurkzrY6dzKjJ6LpCwyFzDHURKCf9WL6hdNeoO9mQA1--P_Az9lzwC5HXazuY8UKUTdc-YBuh6q5oG9U9ZBvOhSxELeUZexLjd855WVfqMTuTXaNUhjbs126YPRmdaBojTA7SLcJuTBjG05n28EWjJZ0CGbhefKLZI9wYj2GKFOEmLfYAl2FaZjDZgWNcIphAWUEa3BRg_ifQZkkIlmLEgXICWsDR4Hyr_TQc0FOi-AY0rOF3ZBF-ak_2NEI8Zj1lj5z2EZ_d7-fs24f3X7cfi6vPl7vt26vC5Ee2RVWitI0pW2FV09W8cY7vJZaVNQYr00lXOlUZXe_z1DrO69YJ63SrdGMx356zl6t3DtOPBWPqB4oGvdcjTkvsJS8bXimp6oy-WlGT_yQGdP0caNDh0AveHyvqjxX1p4oy_OLeu-wHtH_RP51kQKzAHXk8_EfVv7veflqlvwGIlqKs</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>2037054246</pqid></control><display><type>article</type><title>Implications of the International Paediatric Multiple Sclerosis Study Group consensus criteria for paediatric acute disseminated encephalomyelitis: a nationwide validation study</title><source>Access via Wiley Online Library</source><source>Wiley Online Library (Open Access Collection)</source><creator>Boesen, Magnus S ; Blinkenberg, Morten ; Koch‐Henriksen, Nils ; Thygesen, Lau C ; Uldall, Peter V ; Magyari, Melinda ; Born, Alfred P</creator><creatorcontrib>Boesen, Magnus S ; Blinkenberg, Morten ; Koch‐Henriksen, Nils ; Thygesen, Lau C ; Uldall, Peter V ; Magyari, Melinda ; Born, Alfred P</creatorcontrib><description>Aim
The International Paediatric Multiple Sclerosis Study Group (IPMSSG) has proposed criteria for acute disseminated encephalomyelitis (ADEM) not evaluated in clinical practice. Our objective was to assess epidemiological implications of the IPMSSG criteria for ADEM in a cohort study using prospectively collected data.
Method
We identified all diagnosed cases of ADEM in Denmark between 2008 and 2015 from the Danish National Patient Register by International Classification of Diseases 10 codes assigned to acute demyelinating episodes, and we reviewed all medical records to validate ADEM.
Results
We found 52 children up to the age of 18 years with a verified clinical diagnosis of ADEM (incidence rate 0.54/100 000 person‐years; all had abnormal brain magnetic resonance imaging). Only 18 (35%) fulfilled the IPMSSG criteria regarding encephalopathy and polyfocal neurological deficits. Among all 52 children with ADEM, 33 per cent had clinical sequelae after a median follow‐up of 4 years 6 months (range: 10mo–8y 3mo). Surprisingly, none progressed to multiphasic ADEM or multiple sclerosis, but median age at end of follow‐up was only 10 years 9 months (range: 2y–24y 3mo).
Interpretation
Among 52 children with ADEM, none converted to multiphasic ADEM or multiple sclerosis (median follow‐up: 4y 6mo; range: 10mo–8y 3mo). Applying the IPMSSG criteria to all children with a diagnosis of ADEM leaves 65 per cent of the cases without a diagnosis and lowers the incidence rate of paediatric ADEM.
What this paper adds
The incidence of paediatric acute disseminated encephalomyelitis (ADEM) was 0.54 per 100 000 person‐years in children younger than 18 years.
Only 35 per cent of children with ADEM fulfilled the International Paediatric Study Group consensus criteria.
ADEM in clinical practice was primarily based on magnetic resonance imaging findings.
Paediatric neurologists diagnosed ADEM in the absence of encephalopathy.
None of the children with ADEM progressed to multiple sclerosis/multiphasic ADEM during follow‐up.
Resumen
Implicancias en los criterios del consenso del grupo de estudio internacional de esclerosis múltiple pediátrico para la encefalitis aguda diseminada pediátrica: un estudio de validación nacional
Objetivos
El grupo de estudios internacional de esclerosis múltiple pediátrico (IPMSSG) ha propuesto criterios para la encefalitis aguda diseminada (ADEM) no evaluada en la práctica clínica. Nuestro objetivo es evaluar las implicancias epidemiológicas de los criterios de IPMSSG para ADEM en un estudio de cohorte usando datos recolectados de forma prospectiva.
Metodo
Identificamos todos los casos diagnosticados de ADEM en Dinamarca entre el 2008 y el 2015 del registro Nacional de Dinamarca por la Clasificación Internacional de Enfermedades código 10 asignados a episodios agudos de desmielinización, y revisamos todos los registros médicos para validar ADEM.
Resultados
Encontramos 52 niños hasta 18 años de edad con un diagnóstico clínico verificado de ADEM (tasa de incidencia de 0.54/100.000 personas/año; todos tuvieron unas imágenes de resonancia magnética cerebral anormales). Solo 18 (35%) cumplieron con los criterios de IPMSSG en lo que respecta a encefalopatía y déficits neurológicos multifocales. Dentro de los 52 niños con ADEM, 33 por ciento tuvieron secuelas clínicas luego de una mediana de seguimiento de 4 años y 6 meses (rango: 10 meses ‐ 8 años y 3 meses). Sorprendentemente, ninguno progreso a un multifacético ADEM o esclerosis múltiple, pero la edad mediana al final del seguimiento fue de solo 10 años 9 meses (rango: 2 años – 24 años y 3 meses).
Interpretacion
Dentro de los 52 niños con ADEM, ninguno convirtió a un multifacético ADEM o esclerosis múltiple (mediana de seguimiento: 4 años y 6 meses; rango 10 meses‐8 años y 3 meses). Aplicando los criterios de IPMSSG a todos los niños con diagnóstico de ADEM deja un 65 por ciento de los casos sin diagnóstico y más bajas tasas de incidencia de ADEM en pediatría.
Resumo
Implicações do critérios do consenso do Grupo Internacional de Estudos da Esclerose Múltipla pediátrica: um estudo de validação nacional
Objetivo
O Grupo Internacional de Estudos da Esclerose Múltipla Pediátrica (GIEEMP) propôs critérios para a encefalomielite aguda disseminada (EMAD) não avaliada na prática clínica. Nosso objetivo foi avaliar as implicações epidemiológicas dos critérios do GIEEMP para EMAD em uma coorte usando dados coletados prospectivamente.
Método
Identificamos todos os casos diagnosticados de EMAD na Dinamarca ente 2008 e 2015 a partir do Registro Nacional de Pacientes Dinamarqueses por meio dos códigos da Classificação Internacional de Doenças 10 relacionados a episódios desmielinizantes agudos, e revisamos todos os registros médicos para validar a EMAD.
Resultados
Encontramos 52 crianças com idade de até 18 anos com diagnóstico clínico verificado de EMAD (taxa de incidência 0,54/100.000 pessoas por ano; todos tiveram anormalidades no exame de ressonância magnética cerebral). Apenas 18 (35%) atingiram os critérios do GIEEMP para encefalopatia e déficits neurológicos polifocais. Entre as 52 crianças com EMAD, 33 por cento tiveram sequelas clínicas após acompanhamento mediano de 4 anos e 6 meses (variação: 10 m – 8a3m). Surpreendentemente, nenhuma progrediu para EMAD multifásica ou esclerose múltipla, mas a idade mediana no final do acompanhamento foi de apenas 10 anos e 9 meses (variação: 2a‐24a3m).
Interpretação
Entre 52 crianças com EMAD, nenhuma se converteu para EMAD multifásica ou esclerose múltipla (acompanhamento mediano: 4 anos e 6 meses; variação: 10 m – 8a3m). A aplicação dos critérios do GIEEMP para todas as crianças com diagnóstico de EMAD deixa 65% dos casos sem diagnóstico e diminui a taxa de incidência de EMAD pediátrica.
What this paper adds
The incidence of paediatric acute disseminated encephalomyelitis (ADEM) was 0.54 per 100 000 person‐years in children younger than 18 years.
Only 35 per cent of children with ADEM fulfilled the International Paediatric Study Group consensus criteria.
ADEM in clinical practice was primarily based on magnetic resonance imaging findings.
Paediatric neurologists diagnosed ADEM in the absence of encephalopathy.
None of the children with ADEM progressed to multiple sclerosis/multiphasic ADEM during follow‐up.
This article is commented on by Hardy on page 1070 of this issue.
This article's has been translated into Spanish and Portuguese.
Follow the links from the to view the translations.</description><identifier>ISSN: 0012-1622</identifier><identifier>EISSN: 1469-8749</identifier><identifier>DOI: 10.1111/dmcn.13798</identifier><identifier>PMID: 29744874</identifier><language>eng</language><publisher>England</publisher><ispartof>Developmental medicine and child neurology, 2018-11, Vol.60 (11), p.1123-1131</ispartof><rights>2018 Mac Keith Press</rights><rights>2018 Mac Keith Press.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c3658-53e2d7c381d479607ff0b2e35dcce5c92f3f45ca6be5c8f0068f1dfa84a7de2f3</citedby><cites>FETCH-LOGICAL-c3658-53e2d7c381d479607ff0b2e35dcce5c92f3f45ca6be5c8f0068f1dfa84a7de2f3</cites><orcidid>0000-0002-2788-1974</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1111%2Fdmcn.13798$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1111%2Fdmcn.13798$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>314,780,784,1417,1433,27924,27925,45574,45575,46409,46833</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/29744874$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Boesen, Magnus S</creatorcontrib><creatorcontrib>Blinkenberg, Morten</creatorcontrib><creatorcontrib>Koch‐Henriksen, Nils</creatorcontrib><creatorcontrib>Thygesen, Lau C</creatorcontrib><creatorcontrib>Uldall, Peter V</creatorcontrib><creatorcontrib>Magyari, Melinda</creatorcontrib><creatorcontrib>Born, Alfred P</creatorcontrib><title>Implications of the International Paediatric Multiple Sclerosis Study Group consensus criteria for paediatric acute disseminated encephalomyelitis: a nationwide validation study</title><title>Developmental medicine and child neurology</title><addtitle>Dev Med Child Neurol</addtitle><description>Aim
The International Paediatric Multiple Sclerosis Study Group (IPMSSG) has proposed criteria for acute disseminated encephalomyelitis (ADEM) not evaluated in clinical practice. Our objective was to assess epidemiological implications of the IPMSSG criteria for ADEM in a cohort study using prospectively collected data.
Method
We identified all diagnosed cases of ADEM in Denmark between 2008 and 2015 from the Danish National Patient Register by International Classification of Diseases 10 codes assigned to acute demyelinating episodes, and we reviewed all medical records to validate ADEM.
Results
We found 52 children up to the age of 18 years with a verified clinical diagnosis of ADEM (incidence rate 0.54/100 000 person‐years; all had abnormal brain magnetic resonance imaging). Only 18 (35%) fulfilled the IPMSSG criteria regarding encephalopathy and polyfocal neurological deficits. Among all 52 children with ADEM, 33 per cent had clinical sequelae after a median follow‐up of 4 years 6 months (range: 10mo–8y 3mo). Surprisingly, none progressed to multiphasic ADEM or multiple sclerosis, but median age at end of follow‐up was only 10 years 9 months (range: 2y–24y 3mo).
Interpretation
Among 52 children with ADEM, none converted to multiphasic ADEM or multiple sclerosis (median follow‐up: 4y 6mo; range: 10mo–8y 3mo). Applying the IPMSSG criteria to all children with a diagnosis of ADEM leaves 65 per cent of the cases without a diagnosis and lowers the incidence rate of paediatric ADEM.
What this paper adds
The incidence of paediatric acute disseminated encephalomyelitis (ADEM) was 0.54 per 100 000 person‐years in children younger than 18 years.
Only 35 per cent of children with ADEM fulfilled the International Paediatric Study Group consensus criteria.
ADEM in clinical practice was primarily based on magnetic resonance imaging findings.
Paediatric neurologists diagnosed ADEM in the absence of encephalopathy.
None of the children with ADEM progressed to multiple sclerosis/multiphasic ADEM during follow‐up.
Resumen
Implicancias en los criterios del consenso del grupo de estudio internacional de esclerosis múltiple pediátrico para la encefalitis aguda diseminada pediátrica: un estudio de validación nacional
Objetivos
El grupo de estudios internacional de esclerosis múltiple pediátrico (IPMSSG) ha propuesto criterios para la encefalitis aguda diseminada (ADEM) no evaluada en la práctica clínica. Nuestro objetivo es evaluar las implicancias epidemiológicas de los criterios de IPMSSG para ADEM en un estudio de cohorte usando datos recolectados de forma prospectiva.
Metodo
Identificamos todos los casos diagnosticados de ADEM en Dinamarca entre el 2008 y el 2015 del registro Nacional de Dinamarca por la Clasificación Internacional de Enfermedades código 10 asignados a episodios agudos de desmielinización, y revisamos todos los registros médicos para validar ADEM.
Resultados
Encontramos 52 niños hasta 18 años de edad con un diagnóstico clínico verificado de ADEM (tasa de incidencia de 0.54/100.000 personas/año; todos tuvieron unas imágenes de resonancia magnética cerebral anormales). Solo 18 (35%) cumplieron con los criterios de IPMSSG en lo que respecta a encefalopatía y déficits neurológicos multifocales. Dentro de los 52 niños con ADEM, 33 por ciento tuvieron secuelas clínicas luego de una mediana de seguimiento de 4 años y 6 meses (rango: 10 meses ‐ 8 años y 3 meses). Sorprendentemente, ninguno progreso a un multifacético ADEM o esclerosis múltiple, pero la edad mediana al final del seguimiento fue de solo 10 años 9 meses (rango: 2 años – 24 años y 3 meses).
Interpretacion
Dentro de los 52 niños con ADEM, ninguno convirtió a un multifacético ADEM o esclerosis múltiple (mediana de seguimiento: 4 años y 6 meses; rango 10 meses‐8 años y 3 meses). Aplicando los criterios de IPMSSG a todos los niños con diagnóstico de ADEM deja un 65 por ciento de los casos sin diagnóstico y más bajas tasas de incidencia de ADEM en pediatría.
Resumo
Implicações do critérios do consenso do Grupo Internacional de Estudos da Esclerose Múltipla pediátrica: um estudo de validação nacional
Objetivo
O Grupo Internacional de Estudos da Esclerose Múltipla Pediátrica (GIEEMP) propôs critérios para a encefalomielite aguda disseminada (EMAD) não avaliada na prática clínica. Nosso objetivo foi avaliar as implicações epidemiológicas dos critérios do GIEEMP para EMAD em uma coorte usando dados coletados prospectivamente.
Método
Identificamos todos os casos diagnosticados de EMAD na Dinamarca ente 2008 e 2015 a partir do Registro Nacional de Pacientes Dinamarqueses por meio dos códigos da Classificação Internacional de Doenças 10 relacionados a episódios desmielinizantes agudos, e revisamos todos os registros médicos para validar a EMAD.
Resultados
Encontramos 52 crianças com idade de até 18 anos com diagnóstico clínico verificado de EMAD (taxa de incidência 0,54/100.000 pessoas por ano; todos tiveram anormalidades no exame de ressonância magnética cerebral). Apenas 18 (35%) atingiram os critérios do GIEEMP para encefalopatia e déficits neurológicos polifocais. Entre as 52 crianças com EMAD, 33 por cento tiveram sequelas clínicas após acompanhamento mediano de 4 anos e 6 meses (variação: 10 m – 8a3m). Surpreendentemente, nenhuma progrediu para EMAD multifásica ou esclerose múltipla, mas a idade mediana no final do acompanhamento foi de apenas 10 anos e 9 meses (variação: 2a‐24a3m).
Interpretação
Entre 52 crianças com EMAD, nenhuma se converteu para EMAD multifásica ou esclerose múltipla (acompanhamento mediano: 4 anos e 6 meses; variação: 10 m – 8a3m). A aplicação dos critérios do GIEEMP para todas as crianças com diagnóstico de EMAD deixa 65% dos casos sem diagnóstico e diminui a taxa de incidência de EMAD pediátrica.
What this paper adds
The incidence of paediatric acute disseminated encephalomyelitis (ADEM) was 0.54 per 100 000 person‐years in children younger than 18 years.
Only 35 per cent of children with ADEM fulfilled the International Paediatric Study Group consensus criteria.
ADEM in clinical practice was primarily based on magnetic resonance imaging findings.
Paediatric neurologists diagnosed ADEM in the absence of encephalopathy.
None of the children with ADEM progressed to multiple sclerosis/multiphasic ADEM during follow‐up.
This article is commented on by Hardy on page 1070 of this issue.
This article's has been translated into Spanish and Portuguese.
Follow the links from the to view the translations.</description><issn>0012-1622</issn><issn>1469-8749</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2018</creationdate><recordtype>article</recordtype><recordid>eNp9kcuOFCEUhonROO3oxgcwZ2lMagSKurkzrY6dzKjJ6LpCwyFzDHURKCf9WL6hdNeoO9mQA1--P_Az9lzwC5HXazuY8UKUTdc-YBuh6q5oG9U9ZBvOhSxELeUZexLjd855WVfqMTuTXaNUhjbs126YPRmdaBojTA7SLcJuTBjG05n28EWjJZ0CGbhefKLZI9wYj2GKFOEmLfYAl2FaZjDZgWNcIphAWUEa3BRg_ifQZkkIlmLEgXICWsDR4Hyr_TQc0FOi-AY0rOF3ZBF-ak_2NEI8Zj1lj5z2EZ_d7-fs24f3X7cfi6vPl7vt26vC5Ee2RVWitI0pW2FV09W8cY7vJZaVNQYr00lXOlUZXe_z1DrO69YJ63SrdGMx356zl6t3DtOPBWPqB4oGvdcjTkvsJS8bXimp6oy-WlGT_yQGdP0caNDh0AveHyvqjxX1p4oy_OLeu-wHtH_RP51kQKzAHXk8_EfVv7veflqlvwGIlqKs</recordid><startdate>201811</startdate><enddate>201811</enddate><creator>Boesen, Magnus S</creator><creator>Blinkenberg, Morten</creator><creator>Koch‐Henriksen, Nils</creator><creator>Thygesen, Lau C</creator><creator>Uldall, Peter V</creator><creator>Magyari, Melinda</creator><creator>Born, Alfred P</creator><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope><orcidid>https://orcid.org/0000-0002-2788-1974</orcidid></search><sort><creationdate>201811</creationdate><title>Implications of the International Paediatric Multiple Sclerosis Study Group consensus criteria for paediatric acute disseminated encephalomyelitis: a nationwide validation study</title><author>Boesen, Magnus S ; Blinkenberg, Morten ; Koch‐Henriksen, Nils ; Thygesen, Lau C ; Uldall, Peter V ; Magyari, Melinda ; Born, Alfred P</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c3658-53e2d7c381d479607ff0b2e35dcce5c92f3f45ca6be5c8f0068f1dfa84a7de2f3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2018</creationdate><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Boesen, Magnus S</creatorcontrib><creatorcontrib>Blinkenberg, Morten</creatorcontrib><creatorcontrib>Koch‐Henriksen, Nils</creatorcontrib><creatorcontrib>Thygesen, Lau C</creatorcontrib><creatorcontrib>Uldall, Peter V</creatorcontrib><creatorcontrib>Magyari, Melinda</creatorcontrib><creatorcontrib>Born, Alfred P</creatorcontrib><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><jtitle>Developmental medicine and child neurology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Boesen, Magnus S</au><au>Blinkenberg, Morten</au><au>Koch‐Henriksen, Nils</au><au>Thygesen, Lau C</au><au>Uldall, Peter V</au><au>Magyari, Melinda</au><au>Born, Alfred P</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Implications of the International Paediatric Multiple Sclerosis Study Group consensus criteria for paediatric acute disseminated encephalomyelitis: a nationwide validation study</atitle><jtitle>Developmental medicine and child neurology</jtitle><addtitle>Dev Med Child Neurol</addtitle><date>2018-11</date><risdate>2018</risdate><volume>60</volume><issue>11</issue><spage>1123</spage><epage>1131</epage><pages>1123-1131</pages><issn>0012-1622</issn><eissn>1469-8749</eissn><abstract>Aim
The International Paediatric Multiple Sclerosis Study Group (IPMSSG) has proposed criteria for acute disseminated encephalomyelitis (ADEM) not evaluated in clinical practice. Our objective was to assess epidemiological implications of the IPMSSG criteria for ADEM in a cohort study using prospectively collected data.
Method
We identified all diagnosed cases of ADEM in Denmark between 2008 and 2015 from the Danish National Patient Register by International Classification of Diseases 10 codes assigned to acute demyelinating episodes, and we reviewed all medical records to validate ADEM.
Results
We found 52 children up to the age of 18 years with a verified clinical diagnosis of ADEM (incidence rate 0.54/100 000 person‐years; all had abnormal brain magnetic resonance imaging). Only 18 (35%) fulfilled the IPMSSG criteria regarding encephalopathy and polyfocal neurological deficits. Among all 52 children with ADEM, 33 per cent had clinical sequelae after a median follow‐up of 4 years 6 months (range: 10mo–8y 3mo). Surprisingly, none progressed to multiphasic ADEM or multiple sclerosis, but median age at end of follow‐up was only 10 years 9 months (range: 2y–24y 3mo).
Interpretation
Among 52 children with ADEM, none converted to multiphasic ADEM or multiple sclerosis (median follow‐up: 4y 6mo; range: 10mo–8y 3mo). Applying the IPMSSG criteria to all children with a diagnosis of ADEM leaves 65 per cent of the cases without a diagnosis and lowers the incidence rate of paediatric ADEM.
What this paper adds
The incidence of paediatric acute disseminated encephalomyelitis (ADEM) was 0.54 per 100 000 person‐years in children younger than 18 years.
Only 35 per cent of children with ADEM fulfilled the International Paediatric Study Group consensus criteria.
ADEM in clinical practice was primarily based on magnetic resonance imaging findings.
Paediatric neurologists diagnosed ADEM in the absence of encephalopathy.
None of the children with ADEM progressed to multiple sclerosis/multiphasic ADEM during follow‐up.
Resumen
Implicancias en los criterios del consenso del grupo de estudio internacional de esclerosis múltiple pediátrico para la encefalitis aguda diseminada pediátrica: un estudio de validación nacional
Objetivos
El grupo de estudios internacional de esclerosis múltiple pediátrico (IPMSSG) ha propuesto criterios para la encefalitis aguda diseminada (ADEM) no evaluada en la práctica clínica. Nuestro objetivo es evaluar las implicancias epidemiológicas de los criterios de IPMSSG para ADEM en un estudio de cohorte usando datos recolectados de forma prospectiva.
Metodo
Identificamos todos los casos diagnosticados de ADEM en Dinamarca entre el 2008 y el 2015 del registro Nacional de Dinamarca por la Clasificación Internacional de Enfermedades código 10 asignados a episodios agudos de desmielinización, y revisamos todos los registros médicos para validar ADEM.
Resultados
Encontramos 52 niños hasta 18 años de edad con un diagnóstico clínico verificado de ADEM (tasa de incidencia de 0.54/100.000 personas/año; todos tuvieron unas imágenes de resonancia magnética cerebral anormales). Solo 18 (35%) cumplieron con los criterios de IPMSSG en lo que respecta a encefalopatía y déficits neurológicos multifocales. Dentro de los 52 niños con ADEM, 33 por ciento tuvieron secuelas clínicas luego de una mediana de seguimiento de 4 años y 6 meses (rango: 10 meses ‐ 8 años y 3 meses). Sorprendentemente, ninguno progreso a un multifacético ADEM o esclerosis múltiple, pero la edad mediana al final del seguimiento fue de solo 10 años 9 meses (rango: 2 años – 24 años y 3 meses).
Interpretacion
Dentro de los 52 niños con ADEM, ninguno convirtió a un multifacético ADEM o esclerosis múltiple (mediana de seguimiento: 4 años y 6 meses; rango 10 meses‐8 años y 3 meses). Aplicando los criterios de IPMSSG a todos los niños con diagnóstico de ADEM deja un 65 por ciento de los casos sin diagnóstico y más bajas tasas de incidencia de ADEM en pediatría.
Resumo
Implicações do critérios do consenso do Grupo Internacional de Estudos da Esclerose Múltipla pediátrica: um estudo de validação nacional
Objetivo
O Grupo Internacional de Estudos da Esclerose Múltipla Pediátrica (GIEEMP) propôs critérios para a encefalomielite aguda disseminada (EMAD) não avaliada na prática clínica. Nosso objetivo foi avaliar as implicações epidemiológicas dos critérios do GIEEMP para EMAD em uma coorte usando dados coletados prospectivamente.
Método
Identificamos todos os casos diagnosticados de EMAD na Dinamarca ente 2008 e 2015 a partir do Registro Nacional de Pacientes Dinamarqueses por meio dos códigos da Classificação Internacional de Doenças 10 relacionados a episódios desmielinizantes agudos, e revisamos todos os registros médicos para validar a EMAD.
Resultados
Encontramos 52 crianças com idade de até 18 anos com diagnóstico clínico verificado de EMAD (taxa de incidência 0,54/100.000 pessoas por ano; todos tiveram anormalidades no exame de ressonância magnética cerebral). Apenas 18 (35%) atingiram os critérios do GIEEMP para encefalopatia e déficits neurológicos polifocais. Entre as 52 crianças com EMAD, 33 por cento tiveram sequelas clínicas após acompanhamento mediano de 4 anos e 6 meses (variação: 10 m – 8a3m). Surpreendentemente, nenhuma progrediu para EMAD multifásica ou esclerose múltipla, mas a idade mediana no final do acompanhamento foi de apenas 10 anos e 9 meses (variação: 2a‐24a3m).
Interpretação
Entre 52 crianças com EMAD, nenhuma se converteu para EMAD multifásica ou esclerose múltipla (acompanhamento mediano: 4 anos e 6 meses; variação: 10 m – 8a3m). A aplicação dos critérios do GIEEMP para todas as crianças com diagnóstico de EMAD deixa 65% dos casos sem diagnóstico e diminui a taxa de incidência de EMAD pediátrica.
What this paper adds
The incidence of paediatric acute disseminated encephalomyelitis (ADEM) was 0.54 per 100 000 person‐years in children younger than 18 years.
Only 35 per cent of children with ADEM fulfilled the International Paediatric Study Group consensus criteria.
ADEM in clinical practice was primarily based on magnetic resonance imaging findings.
Paediatric neurologists diagnosed ADEM in the absence of encephalopathy.
None of the children with ADEM progressed to multiple sclerosis/multiphasic ADEM during follow‐up.
This article is commented on by Hardy on page 1070 of this issue.
This article's has been translated into Spanish and Portuguese.
Follow the links from the to view the translations.</abstract><cop>England</cop><pmid>29744874</pmid><doi>10.1111/dmcn.13798</doi><tpages>9</tpages><orcidid>https://orcid.org/0000-0002-2788-1974</orcidid><oa>free_for_read</oa></addata></record> |
| fulltext | fulltext |
| identifier | ISSN: 0012-1622 |
| ispartof | Developmental medicine and child neurology, 2018-11, Vol.60 (11), p.1123-1131 |
| issn | 0012-1622 1469-8749 |
| language | eng |
| recordid | cdi_proquest_miscellaneous_2037054246 |
| source | Access via Wiley Online Library; Wiley Online Library (Open Access Collection) |
| title | Implications of the International Paediatric Multiple Sclerosis Study Group consensus criteria for paediatric acute disseminated encephalomyelitis: a nationwide validation study |
| url | https://sfx.bib-bvb.de/sfx_tum?ctx_ver=Z39.88-2004&ctx_enc=info:ofi/enc:UTF-8&ctx_tim=2024-12-24T21%3A19%3A46IST&url_ver=Z39.88-2004&url_ctx_fmt=infofi/fmt:kev:mtx:ctx&rfr_id=info:sid/primo.exlibrisgroup.com:primo3-Article-proquest_cross&rft_val_fmt=info:ofi/fmt:kev:mtx:journal&rft.genre=article&rft.atitle=Implications%20of%20the%20International%20Paediatric%20Multiple%20Sclerosis%20Study%20Group%20consensus%20criteria%20for%20paediatric%20acute%20disseminated%20encephalomyelitis:%20a%20nationwide%20validation%20study&rft.jtitle=Developmental%20medicine%20and%20child%20neurology&rft.au=Boesen,%20Magnus%20S&rft.date=2018-11&rft.volume=60&rft.issue=11&rft.spage=1123&rft.epage=1131&rft.pages=1123-1131&rft.issn=0012-1622&rft.eissn=1469-8749&rft_id=info:doi/10.1111/dmcn.13798&rft_dat=%3Cproquest_cross%3E2037054246%3C/proquest_cross%3E%3Curl%3E%3C/url%3E&disable_directlink=true&sfx.directlink=off&sfx.report_link=0&rft_id=info:oai/&rft_pqid=2037054246&rft_id=info:pmid/29744874&rfr_iscdi=true |