Silent findings: Examination of asymptomatic demyelination in a pediatric US cohort
•Children may present with imaging findings of demyelination.•In our cohort, ⅓ had a clinical attack and ¾ developed new MRI activity.•Predictors for disease activity may include sex and juxtacortical lesion count.•T1 hypointense and infratentorial lesions may predict delayed progression.•Current MS...
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| Veröffentlicht in: | Multiple sclerosis and related disorders 2023-03, Vol.71, p.104573-104573, Article 104573 |
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| creator | Bhise, Vikram Waltz, Michael Casper, T. Charles Aaen, Gregory Benson, Leslie Chitnis, Tanuja Gorman, Mark Goyal, Manu S. Wheeler, Yolanda Lotze, Timothy Mar, Soe Rensel, Mary Abrams, Aaron Rodriguez, Moses Rose, John Schreiner, Teri Shukla, Nikita Waubant, Emmanuelle Weinstock-Guttman, Bianca Ness, Jayne Krupp, Lauren Mendelt-Tillema, Jan |
| description | •Children may present with imaging findings of demyelination.•In our cohort, ⅓ had a clinical attack and ¾ developed new MRI activity.•Predictors for disease activity may include sex and juxtacortical lesion count.•T1 hypointense and infratentorial lesions may predict delayed progression.•Current MS and RIS diagnostic criteria may not be sufficient predictive tools.
Limited data is available on children with evidence of silent central nervous system demyelination on MRI. We sought to characterize the population in a US cohort and identify predictors of clinical and radiologic outcomes.
We identified 56 patients such patients who presented with incidental MRI findings suspect for demyelination, enrolled through our US Network of Pediatric Multiple Sclerosis Centers, and conducted a retrospective review of 38 patients with MR images, and examined risk factors for development of first clinical event or new MRI activity. MRI were rated based on published MS and radiologically isolated syndrome (RIS) imaging diagnostic criteria.
One-third had a clinical attack and ¾ developed new MRI activity over a mean follow-up time of 3.7 years. Individuals in our cohort shared similar demographics to those with clinically definite pediatric-onset MS. We show that sex, presence of infratentorial lesions, T1 hypointense lesions, juxtacortical lesion count, and callosal lesions were predictors of disease progression. Interestingly, the presence of T1 hypointense and infratentorial lesions typically associated with worse outcomes were instead predictive of delayed disease progression on imaging in subgroup analysis. Additionally, currently utilized diagnostic criteria (both McDonald 2017 and RIS criteria) did not provide statistically significant benefit in risk stratification.
Our findings underscore the need for further study to determine if criteria currently used for pediatric patients with purely radiographic evidence of demyelination are sufficient. |
| doi_str_mv | 10.1016/j.msard.2023.104573 |
| format | Article |
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Limited data is available on children with evidence of silent central nervous system demyelination on MRI. We sought to characterize the population in a US cohort and identify predictors of clinical and radiologic outcomes.
We identified 56 patients such patients who presented with incidental MRI findings suspect for demyelination, enrolled through our US Network of Pediatric Multiple Sclerosis Centers, and conducted a retrospective review of 38 patients with MR images, and examined risk factors for development of first clinical event or new MRI activity. MRI were rated based on published MS and radiologically isolated syndrome (RIS) imaging diagnostic criteria.
One-third had a clinical attack and ¾ developed new MRI activity over a mean follow-up time of 3.7 years. Individuals in our cohort shared similar demographics to those with clinically definite pediatric-onset MS. We show that sex, presence of infratentorial lesions, T1 hypointense lesions, juxtacortical lesion count, and callosal lesions were predictors of disease progression. Interestingly, the presence of T1 hypointense and infratentorial lesions typically associated with worse outcomes were instead predictive of delayed disease progression on imaging in subgroup analysis. Additionally, currently utilized diagnostic criteria (both McDonald 2017 and RIS criteria) did not provide statistically significant benefit in risk stratification.
Our findings underscore the need for further study to determine if criteria currently used for pediatric patients with purely radiographic evidence of demyelination are sufficient.</description><identifier>ISSN: 2211-0348</identifier><identifier>EISSN: 2211-0356</identifier><identifier>DOI: 10.1016/j.msard.2023.104573</identifier><identifier>PMID: 36871372</identifier><language>eng</language><publisher>Netherlands: Elsevier B.V</publisher><subject>Autoimmune Diseases of the Nervous System ; Child ; Demyelinating Diseases - diagnostic imaging ; Demyelinating Diseases - epidemiology ; Demyelination ; Disease Progression ; Humans ; Magnetic Resonance Imaging ; MRI ; Multiple Sclerosis - diagnostic imaging ; Multiple Sclerosis - epidemiology ; Pediatric multiple sclerosis ; Radiologically isolated syndrome ; Retrospective Studies ; Risk factors</subject><ispartof>Multiple sclerosis and related disorders, 2023-03, Vol.71, p.104573-104573, Article 104573</ispartof><rights>2023 Elsevier B.V.</rights><rights>Copyright © 2023 Elsevier B.V. All rights reserved.</rights><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c359t-c3ac0f70b9481fc257337285b73d01f0ad37f8d99b2753c6c1f1781c6776365c3</citedby><cites>FETCH-LOGICAL-c359t-c3ac0f70b9481fc257337285b73d01f0ad37f8d99b2753c6c1f1781c6776365c3</cites><orcidid>0000-0003-3304-5523</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,776,780,27901,27902</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/36871372$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Bhise, Vikram</creatorcontrib><creatorcontrib>Waltz, Michael</creatorcontrib><creatorcontrib>Casper, T. Charles</creatorcontrib><creatorcontrib>Aaen, Gregory</creatorcontrib><creatorcontrib>Benson, Leslie</creatorcontrib><creatorcontrib>Chitnis, Tanuja</creatorcontrib><creatorcontrib>Gorman, Mark</creatorcontrib><creatorcontrib>Goyal, Manu S.</creatorcontrib><creatorcontrib>Wheeler, Yolanda</creatorcontrib><creatorcontrib>Lotze, Timothy</creatorcontrib><creatorcontrib>Mar, Soe</creatorcontrib><creatorcontrib>Rensel, Mary</creatorcontrib><creatorcontrib>Abrams, Aaron</creatorcontrib><creatorcontrib>Rodriguez, Moses</creatorcontrib><creatorcontrib>Rose, John</creatorcontrib><creatorcontrib>Schreiner, Teri</creatorcontrib><creatorcontrib>Shukla, Nikita</creatorcontrib><creatorcontrib>Waubant, Emmanuelle</creatorcontrib><creatorcontrib>Weinstock-Guttman, Bianca</creatorcontrib><creatorcontrib>Ness, Jayne</creatorcontrib><creatorcontrib>Krupp, Lauren</creatorcontrib><creatorcontrib>Mendelt-Tillema, Jan</creatorcontrib><creatorcontrib>U.S. Network of Pediatric Multiple Sclerosis Centers</creatorcontrib><title>Silent findings: Examination of asymptomatic demyelination in a pediatric US cohort</title><title>Multiple sclerosis and related disorders</title><addtitle>Mult Scler Relat Disord</addtitle><description>•Children may present with imaging findings of demyelination.•In our cohort, ⅓ had a clinical attack and ¾ developed new MRI activity.•Predictors for disease activity may include sex and juxtacortical lesion count.•T1 hypointense and infratentorial lesions may predict delayed progression.•Current MS and RIS diagnostic criteria may not be sufficient predictive tools.
Limited data is available on children with evidence of silent central nervous system demyelination on MRI. We sought to characterize the population in a US cohort and identify predictors of clinical and radiologic outcomes.
We identified 56 patients such patients who presented with incidental MRI findings suspect for demyelination, enrolled through our US Network of Pediatric Multiple Sclerosis Centers, and conducted a retrospective review of 38 patients with MR images, and examined risk factors for development of first clinical event or new MRI activity. MRI were rated based on published MS and radiologically isolated syndrome (RIS) imaging diagnostic criteria.
One-third had a clinical attack and ¾ developed new MRI activity over a mean follow-up time of 3.7 years. Individuals in our cohort shared similar demographics to those with clinically definite pediatric-onset MS. We show that sex, presence of infratentorial lesions, T1 hypointense lesions, juxtacortical lesion count, and callosal lesions were predictors of disease progression. Interestingly, the presence of T1 hypointense and infratentorial lesions typically associated with worse outcomes were instead predictive of delayed disease progression on imaging in subgroup analysis. Additionally, currently utilized diagnostic criteria (both McDonald 2017 and RIS criteria) did not provide statistically significant benefit in risk stratification.
Our findings underscore the need for further study to determine if criteria currently used for pediatric patients with purely radiographic evidence of demyelination are sufficient.</description><subject>Autoimmune Diseases of the Nervous System</subject><subject>Child</subject><subject>Demyelinating Diseases - diagnostic imaging</subject><subject>Demyelinating Diseases - epidemiology</subject><subject>Demyelination</subject><subject>Disease Progression</subject><subject>Humans</subject><subject>Magnetic Resonance Imaging</subject><subject>MRI</subject><subject>Multiple Sclerosis - diagnostic imaging</subject><subject>Multiple Sclerosis - epidemiology</subject><subject>Pediatric multiple sclerosis</subject><subject>Radiologically isolated syndrome</subject><subject>Retrospective Studies</subject><subject>Risk factors</subject><issn>2211-0348</issn><issn>2211-0356</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2023</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNp9kMlOwzAQhi0EolXpEyChHLm0eElsB4kDqsoiVeJQerYcL-AqiYOdIvr2uHQ5MgePNfP_Hs8HwDWCUwQRvVtPmyiDnmKISarkBSNnYIgxQhNICnp-uud8AMYxrmEKWqCcokswIJQzRBgeguXS1abtM-ta7dqPeJ_Nf2TjWtk732beZjJum673TSqoTJtma-pj17WZzDqjnexDaq6WmfKfPvRX4MLKOprxIY_A6mn-PnuZLN6eX2ePi4kiRdmnUypoGazKnCOrcFohfYkXFSMaIgulJsxyXZYVZgVRVCGLGEeKMkYJLRQZgdv9u13wXxsTe9G4qExdy9b4TRSYccI4zwlJUrKXquBjDMaKLrhGhq1AUOyAirX4Ayp2QMUeaHLdHAZsqsbok-eILwke9gKT1vx2JoionGlVYhKM6oX27t8Bv-KRhtw</recordid><startdate>202303</startdate><enddate>202303</enddate><creator>Bhise, Vikram</creator><creator>Waltz, Michael</creator><creator>Casper, T. 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Charles</au><au>Aaen, Gregory</au><au>Benson, Leslie</au><au>Chitnis, Tanuja</au><au>Gorman, Mark</au><au>Goyal, Manu S.</au><au>Wheeler, Yolanda</au><au>Lotze, Timothy</au><au>Mar, Soe</au><au>Rensel, Mary</au><au>Abrams, Aaron</au><au>Rodriguez, Moses</au><au>Rose, John</au><au>Schreiner, Teri</au><au>Shukla, Nikita</au><au>Waubant, Emmanuelle</au><au>Weinstock-Guttman, Bianca</au><au>Ness, Jayne</au><au>Krupp, Lauren</au><au>Mendelt-Tillema, Jan</au><aucorp>U.S. Network of Pediatric Multiple Sclerosis Centers</aucorp><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Silent findings: Examination of asymptomatic demyelination in a pediatric US cohort</atitle><jtitle>Multiple sclerosis and related disorders</jtitle><addtitle>Mult Scler Relat Disord</addtitle><date>2023-03</date><risdate>2023</risdate><volume>71</volume><spage>104573</spage><epage>104573</epage><pages>104573-104573</pages><artnum>104573</artnum><issn>2211-0348</issn><eissn>2211-0356</eissn><abstract>•Children may present with imaging findings of demyelination.•In our cohort, ⅓ had a clinical attack and ¾ developed new MRI activity.•Predictors for disease activity may include sex and juxtacortical lesion count.•T1 hypointense and infratentorial lesions may predict delayed progression.•Current MS and RIS diagnostic criteria may not be sufficient predictive tools.
Limited data is available on children with evidence of silent central nervous system demyelination on MRI. We sought to characterize the population in a US cohort and identify predictors of clinical and radiologic outcomes.
We identified 56 patients such patients who presented with incidental MRI findings suspect for demyelination, enrolled through our US Network of Pediatric Multiple Sclerosis Centers, and conducted a retrospective review of 38 patients with MR images, and examined risk factors for development of first clinical event or new MRI activity. MRI were rated based on published MS and radiologically isolated syndrome (RIS) imaging diagnostic criteria.
One-third had a clinical attack and ¾ developed new MRI activity over a mean follow-up time of 3.7 years. Individuals in our cohort shared similar demographics to those with clinically definite pediatric-onset MS. We show that sex, presence of infratentorial lesions, T1 hypointense lesions, juxtacortical lesion count, and callosal lesions were predictors of disease progression. Interestingly, the presence of T1 hypointense and infratentorial lesions typically associated with worse outcomes were instead predictive of delayed disease progression on imaging in subgroup analysis. Additionally, currently utilized diagnostic criteria (both McDonald 2017 and RIS criteria) did not provide statistically significant benefit in risk stratification.
Our findings underscore the need for further study to determine if criteria currently used for pediatric patients with purely radiographic evidence of demyelination are sufficient.</abstract><cop>Netherlands</cop><pub>Elsevier B.V</pub><pmid>36871372</pmid><doi>10.1016/j.msard.2023.104573</doi><tpages>1</tpages><orcidid>https://orcid.org/0000-0003-3304-5523</orcidid></addata></record> |
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| ispartof | Multiple sclerosis and related disorders, 2023-03, Vol.71, p.104573-104573, Article 104573 |
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| subjects | Autoimmune Diseases of the Nervous System Child Demyelinating Diseases - diagnostic imaging Demyelinating Diseases - epidemiology Demyelination Disease Progression Humans Magnetic Resonance Imaging MRI Multiple Sclerosis - diagnostic imaging Multiple Sclerosis - epidemiology Pediatric multiple sclerosis Radiologically isolated syndrome Retrospective Studies Risk factors |
| title | Silent findings: Examination of asymptomatic demyelination in a pediatric US cohort |
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