A Rasch Analysis of the Charcot-Marie-Tooth Neuropathy Score (CMTNS) in a Cohort of Charcot-Marie-Tooth Type 1A Patients
| Autor: | Mickaël Guedj, Wenjia Wang, Laurent Magy, Daniel Commenges, Julie Foucquier, Niall P. Murphy, Cécile Proust-Lima, Olivier Blin, Elisabeth Jouve, Shahram Attarian, Viviane Bertrand, Daniel Cohen |
|---|---|
| Přispěvatelé: | Bordeaux population health (BPH), Université de Bordeaux (UB)-Institut de Santé Publique, d'Épidémiologie et de Développement (ISPED)-Institut National de la Santé et de la Recherche Médicale (INSERM), PharNext, Partenaires INRAE, CIC-CPCET, Hôpital de la Timone [CHU - APHM] (TIMONE), Hôpital Dupuytren [CHU Limoges], Génétique Médicale et Génomique Fonctionnelle (GMGF), Aix Marseille Université (AMU)-Assistance Publique - Hôpitaux de Marseille (APHM)- Hôpital de la Timone [CHU - APHM] (TIMONE)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Centre National de la Recherche Scientifique (CNRS) |
| Jazyk: | angličtina |
| Rok vydání: | 2017 |
| Předmět: |
Male
Psychometrics Peripheral neuropathy Physiology Action Potentials lcsh:Medicine Disease Drug research and development Severity of Illness Index Chi Square Tests Cohort Studies 0302 clinical medicine Clinical trials Mathematical and Statistical Techniques 0504 sociology Charcot-Marie-Tooth Disease Outcome Assessment Health Care Medicine and Health Sciences Ethnicities French People Young adult lcsh:Science Musculoskeletal System Multidisciplinary 05 social sciences Middle Aged Electrophysiology Arms Neurology Cohort Physical Sciences Legs Female Anatomy Phase II clinical investigation Statistics (Mathematics) Cohort study Research Article Genetic diseases Adult medicine.medical_specialty congenital hereditary and neonatal diseases and abnormalities Adolescent Neurophysiology Membrane Potential 03 medical and health sciences Young Adult Clinical Trials Phase II as Topic Internal medicine Severity of illness medicine Humans Clinical genetics Statistical Methods Statistical Hypothesis Testing Aged Pharmacology Rasch model business.industry [SCCO.NEUR]Cognitive science/Neuroscience Limbs (Anatomy) lcsh:R 050401 social sciences methods Biology and Life Sciences Models Theoretical Neuropathy Clinical trial Research and analysis methods Clinical medicine People and Places Population Groupings lcsh:Q business 030217 neurology & neurosurgery Mathematics Neuroscience |
| Zdroj: | PLoS ONE PLoS ONE, 2017, 12 (1), pp.e0169878. ⟨10.1371/journal.pone.0169878⟩ PLoS ONE, Vol 12, Iss 1, p e0169878 (2017) |
| ISSN: | 1932-6203 |
| DOI: | 10.1371/journal.pone.0169878⟩ |
| Popis: | International audience; The Charcot-Marie-Tooth Neuropathy Score (CMTNS) was developed as a main efficacy endpoint for application in clinical trials of Charcot-Marie-Tooth disease type 1A (CMT1A). However, the sensitivity of the CMTNS for measuring disease severity and progression in CMT1A patients has been questioned. Here, we applied a Rasch analysis in a French cohort of patients to evaluate the psychometrical properties of the CMTNS. Overall, our analysis supports the validity of the CMTNS for application to CMT1A patients though with some limitations such as certain items of the CMTNS being more suitable for moderate to severe forms of the disease, and some items being disordered. We suggest that additional items and/or categories be considered to better assess mild-to-moderate patients. |
| Databáze: | OpenAIRE |
| Externí odkaz: |
|