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2020
Jonathan D Santoro et al. Neurology.
Objective: To characterize disease severity and distribution
of disability in pediatric-onset multiple sclerosis (POMS) and to develop an
optimized modeling scale for measuring disability we performed a multi-center
retrospective analysis of disability scores in 873 persons with POMS over time
and compared this to previously published data in adults with multiple
sclerosis (MS).
of disability in pediatric-onset multiple sclerosis (POMS) and to develop an
optimized modeling scale for measuring disability we performed a multi-center
retrospective analysis of disability scores in 873 persons with POMS over time
and compared this to previously published data in adults with multiple
sclerosis (MS).
Methods: Retrospective analysis of prospectively collected
data collected from twelve centers of the U.S. Network of Pediatric MS Centers.
Patients were stratified by the number of years from first symptoms of MS to
EDSS assessment and an MS severity score (Ped-MSSS) was calculated per criteria
developed by Roxburgh et al in 2005.
data collected from twelve centers of the U.S. Network of Pediatric MS Centers.
Patients were stratified by the number of years from first symptoms of MS to
EDSS assessment and an MS severity score (Ped-MSSS) was calculated per criteria
developed by Roxburgh et al in 2005.
Results: In total, 873 patients were evaluated. In our
cohort, 52%, 19.4%, and 1.5% of all patients at any time point reached an EDSS
of 2.0, 3.0 and 6.0. Comparison of our Ped-MSSS scores and previously published
adult MSSS scores showed slower progression of Ped-MSSS with increasing gaps
between higher EDSS score and years after diagnosis. Decile scores in our POMS
cohort for EDSS of 2.0, 3.0, and 6.0 were 8.00/9.46/9.94 7.86/9.39/9.91 and
7.32/9.01/9.86 at 2, 5 and 10 years respectively. Notable predictors of disease
progression in both EDSS and Ped-MSSS models were “ever having a motor
relapse”, and EDSS at year 1. Symbol Digit Modalities Test (SDMT) scores
were inversely correlated with duration of disease activity and cerebral
functional score.
cohort, 52%, 19.4%, and 1.5% of all patients at any time point reached an EDSS
of 2.0, 3.0 and 6.0. Comparison of our Ped-MSSS scores and previously published
adult MSSS scores showed slower progression of Ped-MSSS with increasing gaps
between higher EDSS score and years after diagnosis. Decile scores in our POMS
cohort for EDSS of 2.0, 3.0, and 6.0 were 8.00/9.46/9.94 7.86/9.39/9.91 and
7.32/9.01/9.86 at 2, 5 and 10 years respectively. Notable predictors of disease
progression in both EDSS and Ped-MSSS models were “ever having a motor
relapse”, and EDSS at year 1. Symbol Digit Modalities Test (SDMT) scores
were inversely correlated with duration of disease activity and cerebral
functional score.
Conclusions: Persons with POMS exhibit lower EDSS scores
compared to persons with adult-onset MS. Use of a Ped-MSSS model may provide an
alternative to EDSS scoring in clinical assessment of disease severity and
disability accrual.
compared to persons with adult-onset MS. Use of a Ped-MSSS model may provide an
alternative to EDSS scoring in clinical assessment of disease severity and
disability accrual.
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