Prognostic indicators in pediatric clinically isolated syndrome

Iaffaldano, Pietro; Simone, Marta; Lucisano, Giuseppe; Ghezzi, Angelo; Coniglio, Gabriella; Morra, Vincenzo Brescia; Salemi, Giuseppe; Patti, Francesco; Lugaresi, Alessandra; Izquierdo, Guillermo; Bergamaschi, Roberto; Cabrera-Gómez, José Antonio; Pozzilli, Carlo; Millefiorini, Enrico; Alroughani, Raed; Boz, Cavit; Pucci, Eugenio; Zimatore, Giovanni Bosco; Sola, Patrizia; Lus, Giacomo; Maimone, Davide; Avolio, Carlo; Cocco, Eleonora; Sajedi, Seyed Aidin; Costantino, Gianfranco; Duquette, Pierre; Shaygannejad, Vahid; Petersen, Thor; Bolanos, R. Fernandez; Paolicelli, Damiano; Tortorella, Carla; Spelman, Tim; Margari, Lucia; Amato, Maria Pia; Comi, Giancarlo; Butzkueven, Helmut; Trojano, María

doi:10.1002/ana.24938

Cited by 41 publications

(26 citation statements)

References 70 publications

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“…This finding suggests that POMS in the early adulthood and with no or minimal disability show already damage along a WM tract clinically eloquent for ambulation, which represents the major contributor of the disability score (EDSS) in MS. Natural history studies showed that POMS reach irreversible disability and progressive course at a much younger age (i.e., 10 years earlier) than age-matched AOMS patients ( 8 , 9 ). A recent study identified positive and negative prognostic clinical factors for the occurrence of a second clinical attack in pediatric clinically isolated syndrome and disability worsening in POMS ( 36 ). Our results may have a sort of “predictive” meaning for long-term clinical outcome of POMS patients.…”

Section: Discussionmentioning

confidence: 99%

Pronounced Structural and Functional Damage in Early Adult Pediatric-Onset Multiple Sclerosis with No or Minimal Clinical Disability

et al. 2017

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Pediatric-onset multiple sclerosis (POMS) may represent a model of vulnerability to damage occurring during a period of active maturation of the human brain. Whereas adaptive mechanisms seem to take place in the POMS brain in the short-medium term, natural history studies have shown that these patients reach irreversible disability, despite slower progression, at a significantly younger age than adult-onset MS (AOMS) patients. We tested for the first time whether significant brain alterations already occurred in POMS patients in their early adulthood and with no or minimal disability (n = 15) in comparison with age- and disability-matched AOMS patients (n = 14) and to normal controls (NC, n = 20). We used a multimodal MRI approach by modeling, using FSL, voxelwise measures of microstructural integrity of white matter tracts and gray matter volumes with those of intra- and internetwork functional connectivity (FC) (analysis of variance, p ≤ 0.01, corrected for multiple comparisons across space). POMS patients showed, when compared with both NC and AOMS patients, altered measures of diffusion tensor imaging (reduced fractional anisotropy and/or increased diffusivities) and higher probability of lesion occurrence in a clinically eloquent region for physical disability such as the posterior corona radiata. In addition, POMS patients showed, compared with the other two groups, reduced long-range FC, assessed from resting functional MRI, between default mode network and secondary visual network, whose interaction subserves important cognitive functions such as spatial attention and visual learning. Overall, this pattern of structural damage and brain connectivity disruption in early adult POMS patients with no or minimal clinical disability might explain their unfavorable clinical outcome in the long term.

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Section: Discussionmentioning

confidence: 99%

Pronounced Structural and Functional Damage in Early Adult Pediatric-Onset Multiple Sclerosis with No or Minimal Clinical Disability

et al. 2017

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“…D uring the last decades, pediatric multiple sclerosis (MS; ie, clinical onset before age 18 years) has been increasingly recognized, representing from 3 to 10% of the total MS population. [1][2][3] However, only a few longitudinal studies 4,5 have been conducted in these patients.…”

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confidence: 99%

Early Predictors of 9‐Year Disability in Pediatric Multiple Sclerosis

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Bonacchi

Moiola

et al. 2021

Annals of Neurology

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The purpose of this study was to assess early predictors of 9-year disability in pediatric patients with multiple sclerosis. Methods: Clinical and magnetic resonance imaging (MRI) assessments of 123 pediatric patients with multiple sclerosis were obtained at disease onset and after 1 and 2 years. A 9-year clinical follow-up was also performed. Cox proportional hazard and multivariable regression models were used to assess independent predictors of time to first relapse and 9-year outcomes. Results: Time to first relapse was predicted by optic nerve lesions (hazard ratio [HR] = 2.10, p = 0.02) and high-efficacy treatment exposure (HR = 0.31, p = 0.005). Predictors of annualized relapse rate were: at baseline, presence of cerebellar (β = −0.15, p < 0.001), cervical cord lesions (β = 0.16, p = 0.003), and high-efficacy treatment exposure (β = −0.14, p = 0.01); considering also 1-year variables, number of relapses (β = 0.14, p = 0.002), and the previous baseline predictors; considering 2-year variables, time to first relapse (2-year: β = −0.12, p = 0.01) entered, whereas high-efficacy treatment exposure exited the model. Predictors of 9-year disability worsening were: at baseline, presence of optic nerve lesions (odds ratio [OR] = 6.45, p = 0.01); considering 1-year and 2-year variables, Expanded Disability Status Scale (EDSS) changes (1-year: OR = 26.05, p < 0.001; 2-year: OR = 16.38, p = 0.02), and ≥ 2 new T2-lesions in 2 years (2-year: OR = 4.91, p = 0.02). Predictors of higher 9-year EDSS score were: at baseline, EDSS score (β = 0.58, p < 0.001), presence of brainstem lesions (β = 0.31, p = 0.04), and number of cervical cord lesions (β = 0.22, p = 0.05); considering 1-year and 2-year variables, EDSS changes (1-year: β = 0.79, p < 0.001; 2-year: β = 0.55, p < 0.001), and ≥ 2 new T2-lesions (1-year: β = 0.28, p = 0.03; 2-year: β = 0.35, p = 0.01). Interpretation: A complete baseline MRI assessment and an accurate clinical and MRI monitoring during the first 2 years of disease contribute to predict 9-year prognosis in pediatric patients with multiple sclerosis.

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“…Optic neuritis is the most common CIS presentation in pediatrics, followed by transverse myelitis and brainstem syndromes [41]. In a cohort of 770 patients with pediatric CIS who were followed for 10 years to assess the risk of conversion to MS, female gender and multifocal symptoms at onset were risk factors for the occurrence of a second attack [42]. In pediatric optic neuritis, rates of conversion to MS range from 13.8-32% [43,44].…”

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Current Advances in Pediatric Onset Multiple Sclerosis

et al. 2020

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Multiple sclerosis (MS) is an autoimmune inflammatory disease affecting the central nervous system leading to demyelination. MS in the pediatric population is rare, but has been shown to lead to significant disability over the duration of the disease. As we have learned more about pediatric MS, there has been a development of improved diagnostic criteria leading to earlier diagnosis, earlier initiation of disease-modifying therapies (DMT), and an increasing number of DMT used in the treatment of pediatric MS. Over time, treatment with DMT has trended towards the initiation of higher efficacy treatment at time of diagnosis to help prevent further disease progression and accrual of disability over time, and there is evidence in current literature that supports this change in treatment patterns. In this review, we discuss the current knowledge in diagnosis, treatment, and clinical outcomes in pediatric MS.

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Prognostic indicators in pediatric clinically isolated syndrome

Abstract: This work represents a step forward in identifying predictors of unfavorable course in pCIS and POMS and supports a protective effect of early DMD treatment in preventing MS development and disability accumulation in this population. Ann Neurol 2017;81:729-739.

Cited by 41 publications

References 70 publications

Pronounced Structural and Functional Damage in Early Adult Pediatric-Onset Multiple Sclerosis with No or Minimal Clinical Disability

Pronounced Structural and Functional Damage in Early Adult Pediatric-Onset Multiple Sclerosis with No or Minimal Clinical Disability

Early Predictors of 9‐Year Disability in Pediatric Multiple Sclerosis

Current Advances in Pediatric Onset Multiple Sclerosis

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