A framework for measurement and harmonization of pediatric multiple sclerosis etiologic research studies: The Pediatric MS Tool-Kit

Author:

Magalhaes Sandra1,Banwell Brenda2,Bar-Or Amit3,Fortier Isabel4,Hanwell Heather E5,Lim Ming6,Matt Georg E7,Neuteboom Rinze F8,O’Riordan David L9,Schneider Paul K10,Pugliatti Maura11,Shatenstein Bryna12,Tansey Catherine M13,Wassmer Evangeline14,Wolfson Christina15

Affiliation:

1. Royal Victoria Hospital, Allan Memorial Institute and Neuroepidemiology Research Unit, Research Institute of the McGill University Health Centre, Montreal, QC, Canada/Department of Epidemiology, Biostatistics and Occupational Health, McGill University, Montreal, QC, Canada

2. Division of Neurology, Children’s Hospital of Philadelphia, Philadelphia, PA, USA

3. Center for Neuroinflammation and Experimental Therapeutics and Department of Neurology, Perelman School of Medicine, University of Pennsylvania, Philadelphia, PA, USA

4. Research Institute of the McGill University Health Centre, Montreal, QC, Canada

5. Dalla Lana School of Public Health, The University of Toronto, Toronto, ON, Canada

6. Children’s Neurosciences, Evelina London Children’s Hospital, Guy’s and St Thomas’ NHS Foundations Trust, King’s Health Partners Academic Health Sciences Centre, London, UK/ Faculty of Life Sciences & Medicine, King’s College London, London, UK

7. Department of Psychology, San Diego State University, San Diego, CA, USA

8. Department of Pediatric Neurology, Erasmus MC-Sophia Children’s Hospital, Rotterdam, The Netherlands

9. School of Medicine, University of California, San Francisco, San Francisco, CA, USA

10. Royal Victoria Hospital, Allan Memorial Institute and Neuroepidemiology Research Unit, Research Institute of the McGill University Health Centre, Montreal, QC, Canada

11. Department of Medicine, McGill University, Montreal, QC, Canada/ Unit of Clinical Neurology, Department of Biomedical and Surgical Sciences, University of Ferrara, Ferrara, Italy

12. Département de nutrition, Université de Montréal, Montreal, QC, Canada/ Centre de recherche, Institut universitaire de gériatrie de Montréal, CIUSSS du Centre-Sud-de-l’Île-de-Montréal, Montreal, QC, Canada

13. School of Physical and Occupational Therapy, McGill University, Montreal, QC, Canada

14. Department of Neurology, Birmingham Children’s Hospital, Birmingham, UK

15. Royal Victoria Hospital, Allan Memorial Institute and Neuroepidemiology Research Unit, Research Institute of the McGill University Health Centre, Montreal, QC, Canada/ Department of Epidemiology, Biostatistics and Occupational Health, McGill University, Montreal, QC, Canada/ Department of Medicine, McGill University, Montreal, QC, Canada

Abstract

Background: While studying the etiology of multiple sclerosis (MS) in children has several methodological advantages over studying etiology in adults, studies are limited by small sample sizes. Objective: Using a rigorous methodological process, we developed the Pediatric MS Tool-Kit, a measurement framework that includes a minimal set of core variables to assess etiological risk factors. Methods: We solicited input from the International Pediatric MS Study Group to select three risk factors: environmental tobacco smoke (ETS) exposure, sun exposure, and vitamin D intake. To develop the Tool-Kit, we used a Delphi study involving a working group of epidemiologists, neurologists, and content experts from North America and Europe. Results: The Tool-Kit includes six core variables to measure ETS, six to measure sun exposure, and six to measure vitamin D intake. The Tool-Kit can be accessed online ( www.maelstrom-research.org/mica/network/tool-kit ). Conclusion: The goals of the Tool-Kit are to enhance exposure measurement in newly designed pediatric MS studies and comparability of results across studies, and in the longer term to facilitate harmonization of studies, a methodological approach that can be used to circumvent issues of small sample sizes. We believe the Tool-Kit will prove to be a valuable resource to guide pediatric MS researchers in developing study-specific questionnaire

Funder

National Multiple Sclerosis Society

Multiple Sclerosis International Federation

Publisher

SAGE Publications

Subject

Clinical Neurology,Neurology

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