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Prognostic Indicators in Pediatric Clinically Isolated Syndrome Publisher Pubmed



Iaffaldano P1 ; Simone M2 ; Lucisano G1, 3 ; Ghezzi A4 ; Coniglio G5 ; Brescia Morra V6 ; Salemi G7 ; Patti F8 ; Lugaresi A9, 10 ; Izquierdo G11 ; Bergamaschi R12 ; Cabreragomez JA13 ; Pozzilli C14 ; Millefiorini E15 Show All Authors
Authors
  1. Iaffaldano P1
  2. Simone M2
  3. Lucisano G1, 3
  4. Ghezzi A4
  5. Coniglio G5
  6. Brescia Morra V6
  7. Salemi G7
  8. Patti F8
  9. Lugaresi A9, 10
  10. Izquierdo G11
  11. Bergamaschi R12
  12. Cabreragomez JA13
  13. Pozzilli C14
  14. Millefiorini E15
  15. Alroughani R16
  16. Boz C17
  17. Pucci E18
  18. Zimatore GB19
  19. Sola P20
  20. Lus G21
  21. Maimone D22
  22. Avolio C23
  23. Cocco E24
  24. Sajedi SA25
  25. Costantino G26
  26. Duquette P27
  27. Shaygannejad V28
  28. Petersen T29
  29. Fernandez Bolanos R30
  30. Paolicelli D1
  31. Tortorella C1
  32. Spelman T31, 32
  33. Margari L2
  34. Amato MP33
  35. Comi G34
  36. Butzkueven H31, 32
  37. Trojano M1
Show Affiliations
Authors Affiliations
  1. 1. Department of Basic Medical Sciences, Neurosciences, and Sense Organs, University of Bari Aldo Moro, Bari, Italy
  2. 2. Child Neuropsychiatry Unit, Department of Basic Medical Sciences, Neurosciences, and Sense Organs, University of Bari Aldo Moro, Bari, Italy
  3. 3. Center for Outcomes Research and Clinical Epidemiology, CORESEARCH, Pescara, Italy
  4. 4. Multiple Sclerosis Center, Sant'Antonio Abate Hospital, Gallarate, Italy
  5. 5. Neurology Unit, Madonna delle Grazie Hospital, Matera, Italy
  6. 6. Department of Neurosciences, Reproductive and Odontostomatological Sciences, University of Naples Federico II, Naples, Italy
  7. 7. Department of Clinical Neuroscience, University of Palermo, Palermo, Italy
  8. 8. Department of Advanced Medical and Surgical Sciences and Technologies, Multiple Sclerosis Center, University of Catania, Catania, Italy
  9. 9. Department of Biomedical and Neuro Motor Sciences, University of Bologna, Bologna, Italy
  10. 10. IRCCS Institute of Neurological Science and Bellaria Hospital, Bologna, Italy
  11. 11. Department of Neurology, Virgin of Hope of Macarena University Hospital, Seville, Spain
  12. 12. Interdepartment Multiple Sclerosis Research Center, C. Mondino National Institute of Neurology Foundation, Pavia, Italy
  13. 13. International Center for Neurological Restoration, Havana, Cuba
  14. 14. Multiple Sclerosis Center, Sant'Andrea Hospital, Department of Neurology and Psychiatry, Sapienza University, Rome, Italy
  15. 15. Multiple Sclerosis Center, Umberto I Hospital, Sapienza University, Rome, Italy
  16. 16. Division of Neurology, Department of Medicine, Amiri Hospital, Kuwait City, Kuwait
  17. 17. Karadeniz Technical University, Trabzon, Turkey
  18. 18. Neurology Unit, ASUR Marche Hospital, Macerata, Italy
  19. 19. Operative Unit of Neurology, Dimiccoli General Hospital, Barletta, Italy
  20. 20. Department of Neurosciences, Neurology Unit, University of Modena and Reggio Emilia, Sant'Agostino-Estense Hospital, Modena, Italy
  21. 21. Multiple Sclerosis Center, II Division of Neurology, Department of Clinical and Experimental Medicine, Second University of Naples, Naples, Italy
  22. 22. Multiple Sclerosis Center, Garibaldi-Nesima Hospital, Catania, Italy
  23. 23. Department of Medical and Surgical Sciences, University of Foggia, Foggia, Italy
  24. 24. Department of Public Health, Clinical and Molecular Medicine, University of Cagliari, Cagliari, Italy
  25. 25. Multiple Sclerosis Center, Golestan Hospital, Ahvaz Jundishapur University of Medical Sciences, Ahvaz, Iran
  26. 26. Multiple Sclerosis Center, Ospedali Riuniti, Foggia, Italy
  27. 27. Department of Neurology, Notre Dame Hospital, Montreal, QC, Canada
  28. 28. Neurosciences Research Center and Department of Neurology, Isfahan University of Medical Sciences, Isfahan, Iran
  29. 29. Aarhus University Hospital, Aarhus, Denmark
  30. 30. Virgin of Valme University Hospital, Seville, Spain
  31. 31. Department of Neurology, Box Hill Hospital, Monash University, Melbourne, VIC, Australia
  32. 32. Department of Medicine at Royal Melbourne Hospital, University of Melbourne, Parkville, VIC, Australia
  33. 33. Department of NEUROFARBA, University of Florence, Florence, Italy
  34. 34. Department of Neurology, Vita-Salute San Raffaele University, San Raffaele Scientific Institute, Milan, Italy

Source: Annals of Neurology Published:2017


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. Ann Neurol 2017;81:729–739. © 2017 American Neurological Association
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