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Comparison of Spinal Cord Magnetic Resonance Imaging Features Among Children With Acquired Demyelinating Syndromes

IMPORTANCE: The recognition of magnetic resonance imaging (MRI) features associated with distinct causes of myelitis in children is essential to guide investigations and support diagnostic categorization. OBJECTIVE: To determine the clinical and MRI features and outcomes associated with spinal cord...

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Autores principales: Fadda, Giulia, Alves, Cesar A., O’Mahony, Julia, Castro, Denise A., Yeh, E. Ann, Marrie, Ruth Ann, Arnold, Douglas L., Waters, Patrick, Bar-Or, Amit, Vossough, Arastoo, Banwell, Brenda
Formato: Online Artículo Texto
Lenguaje:English
Publicado: American Medical Association 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8515204/
https://www.ncbi.nlm.nih.gov/pubmed/34643718
http://dx.doi.org/10.1001/jamanetworkopen.2021.28871
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author Fadda, Giulia
Alves, Cesar A.
O’Mahony, Julia
Castro, Denise A.
Yeh, E. Ann
Marrie, Ruth Ann
Arnold, Douglas L.
Waters, Patrick
Bar-Or, Amit
Vossough, Arastoo
Banwell, Brenda
author_facet Fadda, Giulia
Alves, Cesar A.
O’Mahony, Julia
Castro, Denise A.
Yeh, E. Ann
Marrie, Ruth Ann
Arnold, Douglas L.
Waters, Patrick
Bar-Or, Amit
Vossough, Arastoo
Banwell, Brenda
author_sort Fadda, Giulia
collection PubMed
description IMPORTANCE: The recognition of magnetic resonance imaging (MRI) features associated with distinct causes of myelitis in children is essential to guide investigations and support diagnostic categorization. OBJECTIVE: To determine the clinical and MRI features and outcomes associated with spinal cord involvement in pediatric myelin oligodendrocyte glycoprotein antibody–associated disease (MOGAD), multiple sclerosis (MS), and seronegative monophasic myelitis. DESIGN, SETTING, AND PARTICIPANTS: In this cohort study, participants were recruited between 2004 and 2017 through the multicenter Canadian Pediatric Demyelinating Disease Study, which enrolled youth younger than 18 years presenting within 90 days of an acquired demyelinating syndrome. Of the 430 participants recruited, those with lesions on available spine MRI and anti-MOG testing performed on archived samples obtained close to clinical presentation were selected. Participants with poor-quality images and final diagnoses of nondemyelinating disease, anti–aquaporin 4 antibody positivity, and relapsing seronegative myelitis were excluded. Data analysis was performed from December 2019 to November 2020. MAIN OUTCOMES AND MEASURES: Spinal cord involvement was evaluated on 324 MRI sequences, with reviewers blinded to clinical, serological, and brain MRI findings. Associated clinical features and disability scores at 5 years of follow-up were retrieved. Results were compared between groups. RESULTS: A total of 107 participants (median [IQR] age at onset, 11.14 [5.59-13.39] years; 55 girls [51%]) were included in the analyses; 40 children had MOGAD, 21 had MS, and 46 had seronegative myelitis. Longitudinally extensive lesions were very common among children with MOGAD (30 of 40 children [75%]), less common among those with seronegative myelitis (20 of 46 children [43%]), and rare in children with MS (1 of 21 children [5%]). Axial gray matter T2-hyperintensity (ie, the H-sign) was observed in 22 of 35 children (63%) with MOGAD, in 14 of 42 children (33%) with seronegative myelitis, and in none of those with MS. The presence of leptomeningeal enhancement was highly suggestive for MOGAD (22 of 32 children [69%] with MOGAD vs 10 of 38 children [26%] with seronegative myelitis and 1 of 15 children [7%] with MS). Children with MOGAD were more likely to have complete lesion resolution on serial images (14 of 21 children [67%]) compared with those with MS (0 of 13 children). CONCLUSIONS AND RELEVANCE: These findings suggest that several features may help identify children at presentation who are more likely to have myelitis associated with MOGAD. Prominent involvement of gray matter and leptomeningeal enhancement are common in pediatric MOGAD, although the pathological underpinning of these observations requires further study.
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spelling pubmed-85152042021-10-29 Comparison of Spinal Cord Magnetic Resonance Imaging Features Among Children With Acquired Demyelinating Syndromes Fadda, Giulia Alves, Cesar A. O’Mahony, Julia Castro, Denise A. Yeh, E. Ann Marrie, Ruth Ann Arnold, Douglas L. Waters, Patrick Bar-Or, Amit Vossough, Arastoo Banwell, Brenda JAMA Netw Open Original Investigation IMPORTANCE: The recognition of magnetic resonance imaging (MRI) features associated with distinct causes of myelitis in children is essential to guide investigations and support diagnostic categorization. OBJECTIVE: To determine the clinical and MRI features and outcomes associated with spinal cord involvement in pediatric myelin oligodendrocyte glycoprotein antibody–associated disease (MOGAD), multiple sclerosis (MS), and seronegative monophasic myelitis. DESIGN, SETTING, AND PARTICIPANTS: In this cohort study, participants were recruited between 2004 and 2017 through the multicenter Canadian Pediatric Demyelinating Disease Study, which enrolled youth younger than 18 years presenting within 90 days of an acquired demyelinating syndrome. Of the 430 participants recruited, those with lesions on available spine MRI and anti-MOG testing performed on archived samples obtained close to clinical presentation were selected. Participants with poor-quality images and final diagnoses of nondemyelinating disease, anti–aquaporin 4 antibody positivity, and relapsing seronegative myelitis were excluded. Data analysis was performed from December 2019 to November 2020. MAIN OUTCOMES AND MEASURES: Spinal cord involvement was evaluated on 324 MRI sequences, with reviewers blinded to clinical, serological, and brain MRI findings. Associated clinical features and disability scores at 5 years of follow-up were retrieved. Results were compared between groups. RESULTS: A total of 107 participants (median [IQR] age at onset, 11.14 [5.59-13.39] years; 55 girls [51%]) were included in the analyses; 40 children had MOGAD, 21 had MS, and 46 had seronegative myelitis. Longitudinally extensive lesions were very common among children with MOGAD (30 of 40 children [75%]), less common among those with seronegative myelitis (20 of 46 children [43%]), and rare in children with MS (1 of 21 children [5%]). Axial gray matter T2-hyperintensity (ie, the H-sign) was observed in 22 of 35 children (63%) with MOGAD, in 14 of 42 children (33%) with seronegative myelitis, and in none of those with MS. The presence of leptomeningeal enhancement was highly suggestive for MOGAD (22 of 32 children [69%] with MOGAD vs 10 of 38 children [26%] with seronegative myelitis and 1 of 15 children [7%] with MS). Children with MOGAD were more likely to have complete lesion resolution on serial images (14 of 21 children [67%]) compared with those with MS (0 of 13 children). CONCLUSIONS AND RELEVANCE: These findings suggest that several features may help identify children at presentation who are more likely to have myelitis associated with MOGAD. Prominent involvement of gray matter and leptomeningeal enhancement are common in pediatric MOGAD, although the pathological underpinning of these observations requires further study. American Medical Association 2021-10-13 /pmc/articles/PMC8515204/ /pubmed/34643718 http://dx.doi.org/10.1001/jamanetworkopen.2021.28871 Text en Copyright 2021 Fadda G et al. JAMA Network Open. https://creativecommons.org/licenses/by/4.0/This is an open access article distributed under the terms of the CC-BY License.
spellingShingle Original Investigation
Fadda, Giulia
Alves, Cesar A.
O’Mahony, Julia
Castro, Denise A.
Yeh, E. Ann
Marrie, Ruth Ann
Arnold, Douglas L.
Waters, Patrick
Bar-Or, Amit
Vossough, Arastoo
Banwell, Brenda
Comparison of Spinal Cord Magnetic Resonance Imaging Features Among Children With Acquired Demyelinating Syndromes
title Comparison of Spinal Cord Magnetic Resonance Imaging Features Among Children With Acquired Demyelinating Syndromes
title_full Comparison of Spinal Cord Magnetic Resonance Imaging Features Among Children With Acquired Demyelinating Syndromes
title_fullStr Comparison of Spinal Cord Magnetic Resonance Imaging Features Among Children With Acquired Demyelinating Syndromes
title_full_unstemmed Comparison of Spinal Cord Magnetic Resonance Imaging Features Among Children With Acquired Demyelinating Syndromes
title_short Comparison of Spinal Cord Magnetic Resonance Imaging Features Among Children With Acquired Demyelinating Syndromes
title_sort comparison of spinal cord magnetic resonance imaging features among children with acquired demyelinating syndromes
topic Original Investigation
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8515204/
https://www.ncbi.nlm.nih.gov/pubmed/34643718
http://dx.doi.org/10.1001/jamanetworkopen.2021.28871
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