TY - JOUR
T1 - Silent findings
T2 - Examination of asymptomatic demyelination in a pediatric US cohort
AU - U.S. Network of Pediatric Multiple Sclerosis Centers
AU - Bhise, Vikram
AU - Waltz, Michael
AU - Casper, T. Charles
AU - Aaen, Gregory
AU - Benson, Leslie
AU - Chitnis, Tanuja
AU - Gorman, Mark
AU - Goyal, Manu S.
AU - Wheeler, Yolanda
AU - Lotze, Timothy
AU - Mar, Soe
AU - Rensel, Mary
AU - Abrams, Aaron
AU - Rodriguez, Moses
AU - Rose, John
AU - Schreiner, Teri
AU - Shukla, Nikita
AU - Waubant, Emmanuelle
AU - Weinstock-Guttman, Bianca
AU - Ness, Jayne
AU - Krupp, Lauren
AU - Mendelt-Tillema, Jan
N1 - Publisher Copyright:
© 2023 Elsevier B.V.
PY - 2023/3
Y1 - 2023/3
N2 - Background and objectives: 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. Methods: 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. Results: 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. Conclusion: 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.
AB - Background and objectives: 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. Methods: 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. Results: 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. Conclusion: 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.
KW - Demyelination
KW - Disease progression
KW - MRI
KW - Pediatric multiple sclerosis
KW - Radiologically isolated syndrome
KW - Risk factors
UR - http://www.scopus.com/inward/record.url?scp=85149363132&partnerID=8YFLogxK
U2 - 10.1016/j.msard.2023.104573
DO - 10.1016/j.msard.2023.104573
M3 - Article
C2 - 36871372
AN - SCOPUS:85149363132
SN - 2211-0348
VL - 71
JO - Multiple Sclerosis and Related Disorders
JF - Multiple Sclerosis and Related Disorders
M1 - 104573
ER -