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November 2017; 4 (6) ArticleOpen Access

Radiologically isolated syndrome in children

Clinical and radiologic outcomes

Naila Makhani, Christine Lebrun, Aksel Siva, David Brassat, Clarisse Carra Dallière, Jérôme de Seze, Wei Du, Françoise Durand Dubief, Orhun Kantarci, Megan Langille, Sona Narula, Jean Pelletier, Juan Ignacio Rojas, Eugene D. Shapiro, Robert T. Stone, Mar Tintoré, Ugur Uygunoglu, Patrick Vermersch, Evangeline Wassmer, Darin T. Okuda, Daniel Pelletier
First published September 25, 2017, DOI: https://doi.org/10.1212/NXI.0000000000000395
Naila Makhani
Author affiliations are provided at the end of the article.
MD, MPH
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Christine Lebrun
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Aksel Siva
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David Brassat
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Clarisse Carra Dallière
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Jérôme de Seze
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Wei Du
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MS
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Françoise Durand Dubief
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Orhun Kantarci
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Megan Langille
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Sona Narula
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Jean Pelletier
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Juan Ignacio Rojas
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Eugene D. Shapiro
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Robert T. Stone
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Mar Tintoré
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Ugur Uygunoglu
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Patrick Vermersch
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Evangeline Wassmer
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Darin T. Okuda
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Daniel Pelletier
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Citation
Radiologically isolated syndrome in children
Clinical and radiologic outcomes
Naila Makhani, Christine Lebrun, Aksel Siva, David Brassat, Clarisse Carra Dallière, Jérôme de Seze, Wei Du, Françoise Durand Dubief, Orhun Kantarci, Megan Langille, Sona Narula, Jean Pelletier, Juan Ignacio Rojas, Eugene D. Shapiro, Robert T. Stone, Mar Tintoré, Ugur Uygunoglu, Patrick Vermersch, Evangeline Wassmer, Darin T. Okuda, Daniel Pelletier
Neurol Neuroimmunol Neuroinflamm Nov 2017, 4 (6) e395; DOI: 10.1212/NXI.0000000000000395

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Abstract

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.

GLOSSARY

CI=
confidence interval;
DIS=
dissemination in space;
HR=
hazard ratio;
IQR=
interquartile range;
RIS=
radiologically isolated syndrome

Footnotes

  • Coinvestigators are listed at Neurology.org/nn.

  • Funding information and disclosures are provided at the end of the article. Go to Neurology.org/nn for full disclosure forms. The Article Processing Charge was funded by the authors.

  • Supplemental data at Neurology.org/nn

  • Received April 25, 2017.
  • Accepted in final form July 7, 2017.
  • 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), 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.

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Neurology - Neuroimmunology Neuroinflammation: 10 (6)

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