Cargando…

Radiologically isolated syndrome in children: Clinical and radiologic outcomes

OBJECTIVE: To describe clinical and radiologic outcomes of children with incidental findings on neuroimaging suggestive of CNS demyelination (termed “radiologically isolated syndrome” or RIS). METHODS: Clinical and radiologic data were obtained from a historical cohort of children with no symptoms o...

Descripción completa

Detalles Bibliográficos
Autores principales: Makhani, Naila, Lebrun, Christine, Siva, Aksel, Brassat, David, Carra Dallière, Clarisse, de Seze, Jérôme, Du, Wei, Durand Dubief, Françoise, Kantarci, Orhun, Langille, Megan, Narula, Sona, Pelletier, Jean, Rojas, Juan Ignacio, Shapiro, Eugene D., Stone, Robert T., Tintoré, Mar, Uygunoglu, Ugur, Vermersch, Patrick, Wassmer, Evangeline, Okuda, Darin T., Pelletier, Daniel
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Lippincott Williams & Wilkins 2017
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5614726/
https://www.ncbi.nlm.nih.gov/pubmed/28959703
http://dx.doi.org/10.1212/NXI.0000000000000395
_version_ 1783266452586889216
author Makhani, Naila
Lebrun, Christine
Siva, Aksel
Brassat, David
Carra Dallière, Clarisse
de Seze, Jérôme
Du, Wei
Durand Dubief, Françoise
Kantarci, Orhun
Langille, Megan
Narula, Sona
Pelletier, Jean
Rojas, Juan Ignacio
Shapiro, Eugene D.
Stone, Robert T.
Tintoré, Mar
Uygunoglu, Ugur
Vermersch, Patrick
Wassmer, Evangeline
Okuda, Darin T.
Pelletier, Daniel
author_facet Makhani, Naila
Lebrun, Christine
Siva, Aksel
Brassat, David
Carra Dallière, Clarisse
de Seze, Jérôme
Du, Wei
Durand Dubief, Françoise
Kantarci, Orhun
Langille, Megan
Narula, Sona
Pelletier, Jean
Rojas, Juan Ignacio
Shapiro, Eugene D.
Stone, Robert T.
Tintoré, Mar
Uygunoglu, Ugur
Vermersch, Patrick
Wassmer, Evangeline
Okuda, Darin T.
Pelletier, Daniel
author_sort Makhani, Naila
collection PubMed
description OBJECTIVE: To describe clinical and radiologic outcomes of children with incidental findings on neuroimaging suggestive of CNS demyelination (termed “radiologically isolated syndrome” or RIS). METHODS: Clinical and radiologic data were obtained from a historical cohort of children with no symptoms of demyelinating disease who had MRI scans that met the 2010 MRI criteria for dissemination in space for MS. RESULTS: We identified 38 children (27 girls and 11 boys) with RIS now being prospectively followed at 16 sites in 6 countries. The mean follow-up time was 4.8 ± 5.3 years. The most common reason for initial neuroimaging was headache (20/38, 53%). A first clinical event consistent with CNS demyelination occurred in 16/38 children (42%; 95% confidence interval [CI]: 27%–60%) in a median of 2.0 years (interquartile range [IQR] 1.0–4.3 years). Radiologic evolution developed in 23/38 children (61%; 95% CI: 44%–76%) in a median of 1.1 years (IQR 0.5–1.9 years). The presence of ≥2 unique oligoclonal bands in CSF (hazard ratio [HR] 10.9, 95% CI: 1.4–86.2, p = 0.02) and spinal cord lesions on MRI (HR 7.8, 95% CI: 1.4–43.6, p = 0.02) were associated with an increased risk of a first clinical event after adjustment for age and sex. CONCLUSIONS: We describe the clinical characteristics and outcomes of children with incidental MRI findings highly suggestive of CNS demyelination. Children with RIS had a substantial risk of subsequent clinical symptoms and/or radiologic evolution. The presence of oligoclonal bands in CSF and spinal cord lesions on MRI were associated with an increased risk of a first clinical event.
format Online
Article
Text
id pubmed-5614726
institution National Center for Biotechnology Information
language English
publishDate 2017
publisher Lippincott Williams & Wilkins
record_format MEDLINE/PubMed
spelling pubmed-56147262017-09-28 Radiologically isolated syndrome in children: Clinical and radiologic outcomes Makhani, Naila Lebrun, Christine Siva, Aksel Brassat, David Carra Dallière, Clarisse de Seze, Jérôme Du, Wei Durand Dubief, Françoise Kantarci, Orhun Langille, Megan Narula, Sona Pelletier, Jean Rojas, Juan Ignacio Shapiro, Eugene D. Stone, Robert T. Tintoré, Mar Uygunoglu, Ugur Vermersch, Patrick Wassmer, Evangeline Okuda, Darin T. Pelletier, Daniel Neurol Neuroimmunol Neuroinflamm Article OBJECTIVE: To describe clinical and radiologic outcomes of children with incidental findings on neuroimaging suggestive of CNS demyelination (termed “radiologically isolated syndrome” or RIS). METHODS: Clinical and radiologic data were obtained from a historical cohort of children with no symptoms of demyelinating disease who had MRI scans that met the 2010 MRI criteria for dissemination in space for MS. RESULTS: We identified 38 children (27 girls and 11 boys) with RIS now being prospectively followed at 16 sites in 6 countries. The mean follow-up time was 4.8 ± 5.3 years. The most common reason for initial neuroimaging was headache (20/38, 53%). A first clinical event consistent with CNS demyelination occurred in 16/38 children (42%; 95% confidence interval [CI]: 27%–60%) in a median of 2.0 years (interquartile range [IQR] 1.0–4.3 years). Radiologic evolution developed in 23/38 children (61%; 95% CI: 44%–76%) in a median of 1.1 years (IQR 0.5–1.9 years). The presence of ≥2 unique oligoclonal bands in CSF (hazard ratio [HR] 10.9, 95% CI: 1.4–86.2, p = 0.02) and spinal cord lesions on MRI (HR 7.8, 95% CI: 1.4–43.6, p = 0.02) were associated with an increased risk of a first clinical event after adjustment for age and sex. CONCLUSIONS: We describe the clinical characteristics and outcomes of children with incidental MRI findings highly suggestive of CNS demyelination. Children with RIS had a substantial risk of subsequent clinical symptoms and/or radiologic evolution. The presence of oligoclonal bands in CSF and spinal cord lesions on MRI were associated with an increased risk of a first clinical event. Lippincott Williams & Wilkins 2017-09-25 /pmc/articles/PMC5614726/ /pubmed/28959703 http://dx.doi.org/10.1212/NXI.0000000000000395 Text en Copyright © 2017 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of the American Academy of Neurology. This is an open access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivatives License 4.0 (CC BY-NC-ND) (http://creativecommons.org/licenses/by-nc-nd/4.0/) , which permits downloading and sharing the work provided it is properly cited. The work cannot be changed in any way or used commercially without permission from the journal.
spellingShingle Article
Makhani, Naila
Lebrun, Christine
Siva, Aksel
Brassat, David
Carra Dallière, Clarisse
de Seze, Jérôme
Du, Wei
Durand Dubief, Françoise
Kantarci, Orhun
Langille, Megan
Narula, Sona
Pelletier, Jean
Rojas, Juan Ignacio
Shapiro, Eugene D.
Stone, Robert T.
Tintoré, Mar
Uygunoglu, Ugur
Vermersch, Patrick
Wassmer, Evangeline
Okuda, Darin T.
Pelletier, Daniel
Radiologically isolated syndrome in children: Clinical and radiologic outcomes
title Radiologically isolated syndrome in children: Clinical and radiologic outcomes
title_full Radiologically isolated syndrome in children: Clinical and radiologic outcomes
title_fullStr Radiologically isolated syndrome in children: Clinical and radiologic outcomes
title_full_unstemmed Radiologically isolated syndrome in children: Clinical and radiologic outcomes
title_short Radiologically isolated syndrome in children: Clinical and radiologic outcomes
title_sort radiologically isolated syndrome in children: clinical and radiologic outcomes
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5614726/
https://www.ncbi.nlm.nih.gov/pubmed/28959703
http://dx.doi.org/10.1212/NXI.0000000000000395
work_keys_str_mv AT makhaninaila radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT lebrunchristine radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT sivaaksel radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT brassatdavid radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT carradalliereclarisse radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT desezejerome radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT duwei radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT duranddubieffrancoise radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT kantarciorhun radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT langillemegan radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT narulasona radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT pelletierjean radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT rojasjuanignacio radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT shapiroeugened radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT stonerobertt radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT tintoremar radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT uygunogluugur radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT vermerschpatrick radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT wassmerevangeline radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT okudadarint radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes
AT pelletierdaniel radiologicallyisolatedsyndromeinchildrenclinicalandradiologicoutcomes