Radiologically isolated syndrome in children

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. OkudaDaniel Pelletier

Research output: Contribution to journalArticle

11 Citations (Scopus)

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.

Original languageEnglish (US)
Article numbere395
JournalNeurology: Neuroimmunology and NeuroInflammation
Volume4
Issue number6
DOIs
StatePublished - Oct 23 2017

Fingerprint

Demyelinating Diseases
Confidence Intervals
Oligoclonal Bands
Incidental Findings
Neuroimaging
Spinal Cord
Headache
Magnetic Resonance Imaging

ASJC Scopus subject areas

  • Neurology
  • Clinical Neurology

Cite this

Makhani, N., Lebrun, C., Siva, A., Brassat, D., Carra Dallière, C., De Seze, J., ... Pelletier, D. (2017). Radiologically isolated syndrome in children. Neurology: Neuroimmunology and NeuroInflammation, 4(6), [e395]. https://doi.org/10.1212/NXI.0000000000000395

Radiologically isolated syndrome in children. / 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.

In: Neurology: Neuroimmunology and NeuroInflammation, Vol. 4, No. 6, e395, 23.10.2017.

Research output: Contribution to journalArticle

Makhani, N, Lebrun, C, Siva, A, Brassat, D, Carra Dallière, C, De Seze, J, Du, W, Durand Dubief, F, Kantarci, O, Langille, M, Narula, S, Pelletier, J, Rojas, JI, Shapiro, ED, Stone, RT, Tintoré, M, Uygunoglu, U, Vermersch, P, Wassmer, E, Okuda, DT & Pelletier, D 2017, 'Radiologically isolated syndrome in children', Neurology: Neuroimmunology and NeuroInflammation, vol. 4, no. 6, e395. https://doi.org/10.1212/NXI.0000000000000395
Makhani N, Lebrun C, Siva A, Brassat D, Carra Dallière C, De Seze J et al. Radiologically isolated syndrome in children. Neurology: Neuroimmunology and NeuroInflammation. 2017 Oct 23;4(6). e395. https://doi.org/10.1212/NXI.0000000000000395
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. In: Neurology: Neuroimmunology and NeuroInflammation. 2017 ; Vol. 4, No. 6.
@article{a28b174c8c4646a88a6651cd35fe6fdf,
title = "Radiologically isolated syndrome in children",
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.",
author = "Naila Makhani and Christine Lebrun and Aksel Siva and David Brassat and {Carra Dalli{\`e}re}, Clarisse and {De Seze}, J{\'e}r{\^o}me and Wei Du and {Durand Dubief}, Fran{\cc}oise and Orhun Kantarci and Megan Langille and Sona Narula and Jean Pelletier and Rojas, {Juan Ignacio} and Shapiro, {Eugene D.} and Stone, {Robert T.} and Mar Tintor{\'e} and Ugur Uygunoglu and Patrick Vermersch and Evangeline Wassmer and Okuda, {Darin T.} and Daniel Pelletier",
year = "2017",
month = "10",
day = "23",
doi = "10.1212/NXI.0000000000000395",
language = "English (US)",
volume = "4",
journal = "Neurology: Neuroimmunology and NeuroInflammation",
issn = "2332-7812",
publisher = "Lippincott Williams and Wilkins",
number = "6",

}

TY - JOUR

T1 - Radiologically isolated syndrome in children

AU - Makhani, Naila

AU - Lebrun, Christine

AU - Siva, Aksel

AU - Brassat, David

AU - Carra Dallière, Clarisse

AU - De Seze, Jérôme

AU - Du, Wei

AU - Durand Dubief, Françoise

AU - Kantarci, Orhun

AU - Langille, Megan

AU - Narula, Sona

AU - Pelletier, Jean

AU - Rojas, Juan Ignacio

AU - Shapiro, Eugene D.

AU - Stone, Robert T.

AU - Tintoré, Mar

AU - Uygunoglu, Ugur

AU - Vermersch, Patrick

AU - Wassmer, Evangeline

AU - Okuda, Darin T.

AU - Pelletier, Daniel

PY - 2017/10/23

Y1 - 2017/10/23

N2 - 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.

AB - 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.

UR - http://www.scopus.com/inward/record.url?scp=85034032686&partnerID=8YFLogxK

UR - http://www.scopus.com/inward/citedby.url?scp=85034032686&partnerID=8YFLogxK

U2 - 10.1212/NXI.0000000000000395

DO - 10.1212/NXI.0000000000000395

M3 - Article

C2 - 28959703

AN - SCOPUS:85034032686

VL - 4

JO - Neurology: Neuroimmunology and NeuroInflammation

JF - Neurology: Neuroimmunology and NeuroInflammation

SN - 2332-7812

IS - 6

M1 - e395

ER -