6533b837fe1ef96bd12a382c
RESEARCH PRODUCT
Prognostic indicators in pediatric clinically isolated syndrome
Pietro IaffaldanoMarta SimoneGiuseppe LucisanoAngelo GhezziGabriella ConiglioVincenzo Brescia MorraGiuseppe SalemiFrancesco PattiAlessandra LugaresiGuillermo IzquierdoRoberto BergamaschiJose Antonio Cabrera-gomezCarlo PozzilliEnrico MillefioriniRaed AlroughaniCavit BozEugenio PucciGiovanni Bosco ZimatorePatrizia SolaGiacomo LusDavide MaimoneCarlo AvolioEleonora CoccoSeyed Aidin SajediGianfranco CostantinoPierre DuquetteVahid ShaygannejadThor PetersenRicardo Fernández BolañosDamiano PaolicelliCarla TortorellaTim SpelmanLucia MargariMaria Pia AmatoGiancarlo ComiHelmut ButzkuevenMaria TrojanoDaniele SpitaleriMaria Rosa RottoliBonaventura ArditoGerardo IulianoEnrico MontanariEnrico GranieriGioacchino TedeschiAntonio BertolottoFranco GranellaGiancarlo Di BattistaPaolo GalloPaola CavallaPaolo BellantonioFrancesca De RobertisLuca DurelliElio ScarpiniMonica RezzonicoAlessandra ProttiClaudio SolaroFrancesco CoreaAntonio BoscoMarika VianelloMaria Teresa FerròRoberto BalgeraRoberta GrassoGiovanna De LucaDeboah FarinaDaniela TravagliniMaria Di IoiaValeria Di TommasoLuca MancinelliErika PietrolongoRaymond HuppertsMaria Edite RioMurat TerziMichael BarnettMark SleeVincent Van PeschAldo SavinoJeannette Lechner-scottPierre GrammondBhim SinghalCees ZwanikkenMarcela FiolLiliana PatruccoMark PainePamela MccombefromFrancois Grand'maisonsubject
RegistrieMaleMultiple SclerosisAdolescentAdolescent; Age of Onset; Child; Demyelinating Diseases; Female; Follow-Up Studies; Humans; Male; Multiple Sclerosis; Prognosis; Retrospective Studies; Risk Factors; Disease Progression; Registries; Neurology; Neurology (clinical)PrognosiONSET MULTIPLE-SCLEROSISCHILDHOODCHILDRENPARACLINICAL FEATURESDISABILITY PROGRESSIONNOFollow-Up StudieRisk FactorsRetrospective Studieprognostic indicatorsMultiple Sclerosipediatric multiple sclerosis prognosis indicatorsHumansRegistriesAge of OnsetChildOPTIC NEURITISRetrospective StudiesRisk FactorDemyelinating DiseaseNATURAL-HISTORYPrognosismultiple sclerosis clinically isolated syndrome prognostic indicatorsNeurologyTRANSVERSE MYELITISclinically isolated syndromeINTERFERON BETA-1BDisease ProgressionSettore MED/26 - NeurologiaFemaleNeurology (clinical)FOLLOW-UPDemyelinating DiseasesFollow-Up StudiesHumandescription
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. 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, 1.06â1.55; 1.42, 1.10â1.84, respectively), whereas disease-modifying drug (DMD) exposure reduced this risk (HR, 95% CI = 0.75, 0.60â0.95). 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, 0.42â0.83; 0.75, 0.71â0.80, respectively), whereas the occurrence of relapse increased this risk (HR, 95% CI = 5.08, 3.46â7.46). 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. Ann Neurol 2017;81:729â739.
| year | journal | country | edition | language |
|---|---|---|---|---|
| 2017-05-01 |