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Title: Examining the environmental risk factors of progressive-onset and relapsing-onset multiple sclerosis: recruitment challenges, potential bias, and statistical strategies.
Austin Authors: Li, Ying;Saul, Alice;Taylor, Bruce;Ponsonby, Anne-Louise;Simpson-Yap, Steve;Blizzard, Leigh;Broadley, Simon;Lechner-Scott, Jeannette;Karabudak, Rana;Patti, Francesco;Eichau, Sara;Onofrj, Marco;Ozakbas, Serkan;Horakova, Dana;Kubala Havrdova, Eva;Grand'Maison, Francois;Alroughani, Raed;Gerlach, Oliver;Amato, Maria Pia;Altintas, Ayse;Girard, Marc;Duquette, Pierre;Blanco, Yolanda;Ramo-Tello, Cristina;Laureys, Guy;Kalincik, Tomas;Khoury, Samia J;Shaygannejad, Vahid;Etemadifar, Masoud;Singhal, Bhim;Mrabet, Saloua;Foschi, Matteo;Habek, Mario;John, Nevin;Hughes, Stella;McCombe, Pamela;Ampapa, Radek;van der Walt, Anneke;Butzkueven, Helmut;de Gans, Koen;McGuigan, Chris;Oreja-Guevara, Celia;Sa, Maria Jose;Petersen, Thor;Al-Harbi, Talal;Sempere, Angel Perez;Van Wijmeersch, Bart;Grigoriadis, Nikolaos;Prevost, Julie;Gray, Orla;Castillo-Triviño, Tamara;Macdonell, Richard A L ;Lugaresi, Alessandra;Sajedi, Seyed Aidin;van der Mei, Ingrid
Affiliation: Menzies Institute of Medical Research, University of Tasmania, 17 Liverpool Street, Hobart, TAS, 7000, Australia.
Florey Institute for Neuroscience, University of Melbourne, Melbourne, VIC, Australia.
School of Medicine and Dentistry, Griffith University, Brisbane, QLD, Australia.
Hunter Medical Research Institute, University of Newcastle, Callaghan, Australia.
Hacettepe University, Ankara, Turkey.
Department of Medical and Surgical Sciences and Advanced Technologies, GF Ingrassia, Catania, Italy.;UOS Sclerosi Multipla, AOU Policlinico "G Rodloico-San Marco", University of Catania, Catania, Italy.
Hospital Universitario Virgen Macarena, Seville, Spain.
University G. d'Annunzio, Chieti, Italy.
Dokuz Eylul University, Konak, Izmir, Turkey.
Charles University in Prague and General University Hospital, Prague, Czech Republic.
Neuro Rive-Sud, Quebec, Canada.
Amiri Hospital, Sharq, Kuwait.
Zuyderland Medical Center, Sittard-Geleen, The Netherlands.;School for Mental Health and Neuroscience, Maastricht University, Maastricht, The Netherlands.
University of Florence, Florence, Italy.
Department of Neurology and Koc University Research Center for Translational Medicine (KUTTAM), Koc University, School of Medicine, Istanbul, Turkey.
CHUM and Universite de Montreal, Montreal, Canada.
Hospital Clinic de Barcelona, Barcelona, Spain.
Hospital Germans Trias i Pujol, Badalona, Spain.
Universitary Hospital Ghent, Ghent, Belgium.
Department of Neurology, Neroimmunology Centre, Royal Melbourne Hospital, Melbourne, Australia.;Department of Medicine, CORe, University of Melbourne, Melbourne, Australia.
American University of Beirut Medical Center, Beirut, Lebanon.
Isfahan University of Medical Sciences, Isfahan, Iran.
Al Zahra Hospital, Isfahan, Iran.
Bombay Hospital Institute of Medical Sciences, Mumbai, India.
Department of Neurology, LR 18SP03, Clinical Investigation Centre Neurosciences and Mental Health, University Hospital Razi-Manouba, Tunis, Tunisia.;Faculty of Medicine of Tunis, University of Tunis El Manar, 1007, Tunis, Tunisia.
S. Maria delle Croci Hospital, AUSL Romagna, Ravenna, Italy.;Neuroscience Section, Department of Applied Clinical Sciences and Biotechnology, University of L'Aquila, Via Vetoio 1, L'Aquila, Italy.
University Hospital Center Zagreb, Zagreb, Croatia.;University of Zagreb, School of Medicine, Zagreb, Croatia.
Monash Medical Centre, Melbourne, Australia.;Department of Medicine, School of Clinical Sciences, Monash University, Melbourne, Australia.
Royal Victoria Hospital, Belfast, UK.
Royal Brisbane and Women's Hospital, Brisbane, Australia.;University of Queensland, Brisbane, Australia.
Nemocnice Jihlava, Jihlava, Czech Republic.
The Alfred Hospital, Melbourne, Australia.;Central Clinical School, Monash University, Melbourne, Australia.
Monash University, Melbourne, Australia.
Groene Hart Ziekenhuis, Gouda, Netherlands.
St Vincent's University Hospital, Dublin, Ireland.;University College Dublin, Dublin, Ireland.
Hospital Clinico San Carlos, Madrid, Spain.
Centro Hospitalar Universitario de Sao Joao, Porto, Portugal.;Faculty of Health Sciences, University Fernando Pessoa, Porto, Portugal.
Aarhus University Hospital, Aarhus, Denmark.
King Fahad Specialist Hospital-Dammam, Khobar, Saudi Arabia.
Hospital General Universitario de Alicante, Alicante, Spain.
Pelt and Hasselt University, Hasselt, Belgium.
AHEPA University Hospital, Thessaloniki, Greece.
CSSS Saint-Jérôme, Saint-Jerome, Canada.
South Eastern HSC Trust, Belfast, UK.
Hospital Universitario Donostia and IIS Biodonostia, San Sebastián, Spain.
Austin Health
IRCCS Istituto delle Scienze Neurologiche di Bologna, Bologna, Italy.;Dipartimento di Scienze Biomediche e Neuromotorie, Università di Bologna, Bologna, Italy.
Golestan University of Medical Sciences, Gogan, Iran.
The Florey Institute of Neuroscience and Mental Health
Department of Medicine, The University of Melbourne, Melbourne, Australia.
Issue Date: 28-Sep-2023
Date: 2023
Publication information: Journal of Neurology 2023-09-28
Abstract: It is unknown whether the currently known risk factors of multiple sclerosis reflect the etiology of progressive-onset multiple sclerosis (POMS) as observational studies rarely included analysis by type of onset. We designed a case-control study to examine associations between environmental factors and POMS and compared effect sizes to relapse-onset MS (ROMS), which will offer insights into the etiology of POMS and potentially contribute to prevention and intervention practice. This study utilizes data from the Primary Progressive Multiple Sclerosis (PPMS) Study and the Australian Multi-center Study of Environment and Immune Function (the AusImmune Study). This report outlines the conduct of the PPMS Study, whether the POMS sample is representative, and the planned analysis methods. The study includes 155 POMS, 204 ROMS, and 558 controls. The distributions of the POMS were largely similar to Australian POMS patients in the MSBase Study, with 54.8% female, 85.8% POMS born before 1970, mean age of onset of 41.44 ± 8.38 years old, and 67.1% living between 28.9 and 39.4° S. The POMS were representative of the Australian POMS population. There are some differences between POMS and ROMS/controls (mean age at interview: POMS 55 years vs. controls 40 years; sex: POMS 53% female vs. controls 78% female; location of residence: 14.3% of POMS at a latitude ≤ 28.9°S vs. 32.8% in controls), which will be taken into account in the analysis. We discuss the methodological issues considered in the study design, including prevalence-incidence bias, cohort effects, interview bias and recall bias, and present strategies to account for it. Associations between exposures of interest and POMS/ROMS will be presented in subsequent publications.
DOI: 10.1007/s00415-023-11980-z
Journal: Journal of Neurology
PubMed URL: 37768389
ISSN: 1432-1459
Type: Journal Article
Subjects: Bias
Environmental factors
Progressive-onset multiple sclerosis
Subject recruitment
Appears in Collections:Journal articles

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