Prognostic indicators in pediatric clinically isolated syndrome

Giuseppe Salemi, Vahid Shaygannejad, Liliana Patrucco, Vincent Van Pesch, Thor Petersen, Maria Di Ioia, Daniela Travaglini, Murat Terzi, Roberto Balgera, Seyed Aidin Sajedi, Bonaventura Ardito, Pietro Iaffaldano, Tim Spelman, Antonio Bosco, Giovanna De Luca, Giuseppe Lucisano, Marcela Fiol, Valeria Di Tommaso, Pierre Grammond, Erika PietrolongoRoberto Bergamaschi, Marta Simone, Maria Teresa Ferrò, Gerardo Iuliano, Aldo Savino, Jose Antonio Cabrera-Gomez, Raymond Hupperts, Lucia Margari, Luca Mancinelli, Roberta Grasso, Marika Vianello, Marika Vianello, Bhim Singhal, Helmut Butzkueven, Paolo Gallo, Patrizia Sola, Michael Barnett, Enrico Granieri, Pamela Mccombefrom, Raed Alroughani, Elio Scarpini, Elio Scarpini, Maria Edite Rio, Francesco Corea, Raed Alroughani, Marika Vianello, Paolo Bellantonio, Ricardo Fernández Bolaños, Francois Grand'Maison, Alessandra Protti, Cees Zwanikken, Damiano Paolicelli, Daniele Spitaleri, Carlo Avolio, Giacomo Lus, Francesca De Robertis, Jeannette Lechner-Scott, Monica Rezzonico, Francesco Corea, Maria Rosa Rottoli, Paola Cavalla, Vincenzo Brescia Morra, Davide Maimone, Cavit Boz, Giovanni Bosco Zimatore, Franco Granella, Eleonora Cocco, Claudio Solaro, Enrico Millefiorini, Alessandra Lugaresi, Deboah Farina, Pierre Duquette, Maria Trojano, Guillermo Izquierdo, Angelo Ghezzi, Eugenio Pucci, Gianfranco Costantino, Mark Paine, Carlo Pozzilli, Giancarlo Di Battista, Carla Tortorella, Antonio Bertolotto, Enrico Montanari, Francesco Patti, Luca Durelli, Gioacchino Tedeschi, Maria Pia Amato, Giancarlo Comi, Gabriella Coniglio, Mark Slee

Risultato della ricerca: Articlepeer review

21 Citazioni (Scopus)

Abstract

OBJECTIVE:To assess prognostic factors for a second clinical attack and a first disability-worsening event in pediatric clinically isolated syndrome (pCIS) suggestive of multiple sclerosis (MS) patients.METHODS:A cohort of 770 pCIS patients was followed up for at least 10 years. Cox proportional hazard models and Recursive Partitioning and Amalgamation (RECPAM) tree-regression were used to analyze data.RESULTS:In pCIS, female sex and a multifocal onset were risk factors for a second clinical attack (hazard ratio [HR], 95% confidence interval [CI] = 1.28, 1.06-1.55; 1.42, 1.10-1.84, respectively), whereas disease-modifying drug (DMD) exposure reduced this risk (HR, 95% CI = 0.75, 0.60-0.95). After pediatric onset MS (POMS) diagnosis, age at onset younger than 15 years and DMD exposure decreased the risk of a first Expanded Disability Status Scale (EDSS)-worsening event (HR, 95% CI = 0.59, 0.42-0.83; 0.75, 0.71-0.80, respectively), whereas the occurrence of relapse increased this risk (HR, 95% CI = 5.08, 3.46-7.46). An exploratory RECPAM analysis highlighted a significantly higher incidence of a first EDSS-worsening event in patients with multifocal or isolated spinal cord or optic neuritis involvement at onset in comparison to those with an isolated supratentorial or brainstem syndrome. A Cox regression model including RECPAM classes confirmed DMD exposure as the most protective factor against EDSS-worsening events and relapses as the most important risk factor for attaining EDSS worsening.INTERPRETATION: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
Lingua originaleEnglish
pagine (da-a)729-739
Numero di pagine11
RivistaAnnals of Neurology
Volume81
Stato di pubblicazionePublished - 2017

All Science Journal Classification (ASJC) codes

  • Neurology
  • Clinical Neurology

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