Literature DB >> 23509048

International Pediatric MS Study Group Clinical Trials Summit: meeting report.

Tanuja Chitnis1, Marc Tardieu, Maria Pia Amato, Brenda Banwell, Amit Bar-Or, Angelo Ghezzi, Andrew Kornberg, Lauren B Krupp, Daniela Pohl, Kevin Rostasy, Silvia Tenembaum, Emmanuelle Waubant, Evangeline Wassmer.   

Abstract

OBJECTIVE: Pediatric studies for new biological agents are mandated by recent legislation, necessitating careful thought to evaluation of emerging multiple sclerosis (MS) therapies in children with MS. Challenges include a small patient population, the lack of prior randomized clinical trials, and ethical concerns. The goal of this meeting was to assess areas of consensus regarding clinical trial design and outcome measures among academic experts involved in pediatric MS care and research.
METHODS: The Steering Committee of the International Pediatric MS Study Group identified key focus areas for discussion. A total of 69 meeting attendees were assembled, including 35 academic experts. Regulatory and pharmaceutical representatives also attended, and provided input, which informed academic expert consensus decisions.
RESULTS: The academic experts agreed that clinical trials were necessary in pediatric MS to obtain pharmacokinetic, safety and efficacy data, and regulatory approval allowing for greater medication access. The academic experts agreed that relapse was an appropriate primary outcome measure for phase III pediatric trials. An international standardized cognitive battery was identified. The pros and cons of various trial designs were discussed. Guidelines surrounding MRI studies, pharmacokinetics, pharmacodynamics, and registries were developed. The academic experts agreed that given the limited subject pool, a stepwise approach to the launch of clinical trials for the most promising medications is necessary in order to ensure study completion. Alternative approaches could result in unethical exposure of patients to trial conditions without gaining knowledge.
CONCLUSION: Consensus points for conduct of clinical trials in the rare disease pediatric MS were identified amongst a panel of academic experts, informed by regulatory and industry stakeholders.

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Year:  2013        PMID: 23509048      PMCID: PMC3662305          DOI: 10.1212/WNL.0b013e318288694e

Source DB:  PubMed          Journal:  Neurology        ISSN: 0028-3878            Impact factor:   9.910


  27 in total

1.  Paediatric multiple sclerosis and acute disseminated encephalomyelitis in Germany: results of a nationwide survey.

Authors:  Daniela Pohl; Isabell Hennemuth; Rüdiger von Kries; Folker Hanefeld
Journal:  Eur J Pediatr       Date:  2007-01-12       Impact factor: 3.183

2.  MRI quantification of gray and white matter damage in patients with early-onset multiple sclerosis.

Authors:  P Tortorella; M A Rocca; D M Mezzapesa; A Ghezzi; L Lamantia; G Comi; M Filippi
Journal:  J Neurol       Date:  2006-03-06       Impact factor: 4.849

3.  Atypical forms of multiple sclerosis or different phases of a same disease?

Authors:  M Trojano; D Paolicelli; A Bellacosa; A Fuiani; S Cataldi; E Di Monte
Journal:  Neurol Sci       Date:  2004-11       Impact factor: 3.307

4.  Incidence of acquired CNS demyelinating syndromes in a multiethnic cohort of children.

Authors:  A Langer-Gould; J L Zhang; J Chung; Y Yeung; E Waubant; J Yao
Journal:  Neurology       Date:  2011-08-24       Impact factor: 9.910

5.  Early onset multiple sclerosis: a longitudinal study.

Authors:  A Boiko; G Vorobeychik; D Paty; V Devonshire; D Sadovnick
Journal:  Neurology       Date:  2002-10-08       Impact factor: 9.910

6.  MRI correlates of cognitive impairment in childhood-onset multiple sclerosis.

Authors:  C Till; R Ghassemi; B Aubert-Broche; A Kerbrat; D L Collins; S Narayanan; D L Arnold; M Desrocher; J G Sled; B L Banwell
Journal:  Neuropsychology       Date:  2011-05       Impact factor: 3.295

Review 7.  The psychosocial consequences of pediatric multiple sclerosis.

Authors:  W S MacAllister; J R Boyd; N J Holland; M C Milazzo; L B Krupp
Journal:  Neurology       Date:  2007-04-17       Impact factor: 9.910

Review 8.  Consensus definitions proposed for pediatric multiple sclerosis and related disorders.

Authors:  Lauren B Krupp; Brenda Banwell; Silvia Tenembaum
Journal:  Neurology       Date:  2007-04-17       Impact factor: 9.910

9.  Multiple sclerosis therapies in pediatric patients with refractory multiple sclerosis.

Authors:  E Ann Yeh; Emmanuelle Waubant; Lauren B Krupp; Jayne Ness; Tanuja Chitnis; Nancy Kuntz; Murali Ramanathan; Anita Belman; Dorothee Chabas; Mark P Gorman; Moses Rodriguez; John Robert Rinker; Bianca Weinstock-Guttman
Journal:  Arch Neurol       Date:  2010-12-13

10.  Cognitive functioning in children and adolescents with multiple sclerosis.

Authors:  W S MacAllister; A L Belman; M Milazzo; D M Weisbrot; C Christodoulou; W F Scherl; T E Preston; C Cianciulli; L B Krupp
Journal:  Neurology       Date:  2005-04-26       Impact factor: 9.910

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  16 in total

1.  Monocular and binocular low-contrast visual acuity and optical coherence tomography in pediatric multiple sclerosis.

Authors:  Amy T Waldman; Girish Hiremath; Robert A Avery; Amy Conger; Stacy L Pineles; Michael J Loguidice; Lauren S Talman; Kristin M Galetta; Michael J Shumski; James Wilson; E'tona Ford; Amy M Lavery; Darrel Conger; Benjamin M Greenberg; Jonas H Ellenberg; Elliot M Frohman; Laura J Balcer; Peter A Calabresi
Journal:  Mult Scler Relat Disord       Date:  2013-05-01       Impact factor: 4.339

Review 2.  Multiple sclerosis in children: an update on clinical diagnosis, therapeutic strategies, and research.

Authors:  Amy Waldman; Angelo Ghezzi; Amit Bar-Or; Yann Mikaeloff; Marc Tardieu; Brenda Banwell
Journal:  Lancet Neurol       Date:  2014-09       Impact factor: 44.182

Review 3.  Children with multiple sclerosis should not become therapeutic hostages.

Authors:  Klaus Rose; Thomas Müller
Journal:  Ther Adv Neurol Disord       Date:  2016-07-11       Impact factor: 6.570

Review 4.  Outcome measures in relapsing-remitting multiple sclerosis: capturing disability and disease progression in clinical trials.

Authors:  Amy M Lavery; Leonard H Verhey; Amy T Waldman
Journal:  Mult Scler Int       Date:  2014-05-04

Review 5.  Multiple sclerosis: clinical profiling and data collection as prerequisite for personalized medicine approach.

Authors:  Tjalf Ziemssen; Raimar Kern; Katja Thomas
Journal:  BMC Neurol       Date:  2016-08-02       Impact factor: 2.474

6.  Pediatric Multiple Sclerosis Severity Score in a large US cohort.

Authors:  Jonathan D Santoro; Michael Waltz; Greg Aaen; Anita Belman; Leslie Benson; Mark Gorman; Manu S Goyal; Jennifer S Graves; Yolanda Harris; Lauren Krupp; Timothy Lotze; Soe Mar; Manikum Moodley; Jayne Ness; Mary Rensel; Moses Rodriguez; Teri Schreiner; Jan-Mendelt Tillema; Emmanuelle Waubant; Bianca Weinstock-Guttman; Brigitte F Hurtubise; Shelly Roalstad; John Rose; T Charles Casper; Tanuja Chitnis
Journal:  Neurology       Date:  2020-07-20       Impact factor: 9.910

Review 7.  Use of Disease-Modifying Therapies in Pediatric MS.

Authors:  Marta Simone; Tanuja Chitnis
Journal:  Curr Treat Options Neurol       Date:  2016-08       Impact factor: 3.972

8.  Natalizumab in the pediatric MS population: results of the Italian registry.

Authors:  Angelo Ghezzi; Lucia Moiola; Carlo Pozzilli; Vincenzo Brescia-Morra; Paolo Gallo; Luigi Maria Edoardo Grimaldi; Massimo Filippi; Giancarlo Comi G
Journal:  BMC Neurol       Date:  2015-09-25       Impact factor: 2.474

9.  Recommendations and evidence for reporting items in pediatric clinical trial protocols and reports: two systematic reviews.

Authors:  April V P Clyburne-Sherin; Pravheen Thurairajah; Mufiza Z Kapadia; Margaret Sampson; Winnie W Y Chan; Martin Offringa
Journal:  Trials       Date:  2015-09-18       Impact factor: 2.279

Review 10.  Pediatric multiple sclerosis: current concepts and consensus definitions.

Authors:  Joaquin A Pena; Timothy E Lotze
Journal:  Autoimmune Dis       Date:  2013-11-02
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