Walking test outcomes in adults with genetic neuromuscular diseases: A systematic literature review of their measurement properties
2024
Hadouiri, Nawale | Fournel, Isabelle | Thauvin-Robinet, Christel | Jacquin-Piques, Agnès | Ornetti, Paul | Gueugnon, Mathieu | CHU Dijon ; Centre Hospitalier Universitaire de Dijon - Hôpital François Mitterrand (CHU Dijon) | Université Bourgogne Franche-Comté [COMUE] (UBFC) | Centre d'Investigation Clinique 1432 (Dijon) - Epidemiologie Clinique/Essais Cliniques (CIC-EC) ; Université de Bourgogne (UB)-Centre Hospitalier Universitaire de Dijon - Hôpital François Mitterrand (CHU Dijon)-Institut National de la Santé et de la Recherche Médicale (INSERM) | Génétique des anomalies du développement (CTM UMR 1231) (GAD) ; Center for Translational and Molecular medicine [Dijon - UMR1231] (CTM) ; École Pratique des Hautes Études (EPHE) ; Université Paris Sciences et Lettres (PSL)-Université Paris Sciences et Lettres (PSL)-Université de Bourgogne (UB)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Institut Agro Dijon ; Institut national d'enseignement supérieur pour l'agriculture, l'alimentation et l'environnement (Institut Agro)-Institut national d'enseignement supérieur pour l'agriculture, l'alimentation et l'environnement (Institut Agro)-École Pratique des Hautes Études (EPHE) ; Université Paris Sciences et Lettres (PSL)-Université Paris Sciences et Lettres (PSL)-Université de Bourgogne (UB)-Institut National de la Santé et de la Recherche Médicale (INSERM)-Institut Agro Dijon ; Institut national d'enseignement supérieur pour l'agriculture, l'alimentation et l'environnement (Institut Agro)-Institut national d'enseignement supérieur pour l'agriculture, l'alimentation et l'environnement (Institut Agro) | Service de Neurophysiologie Clinique (CHU Dijon) ; Centre Hospitalier Universitaire de Dijon - Hôpital François Mitterrand (CHU Dijon) | Centre des Sciences du Goût et de l'Alimentation [Dijon] (CSGA) ; Université de Bourgogne (UB)-Centre National de la Recherche Scientifique (CNRS)-Institut National de Recherche pour l’Agriculture, l’Alimentation et l’Environnement (INRAE)-Institut Agro Dijon ; Institut national d'enseignement supérieur pour l'agriculture, l'alimentation et l'environnement (Institut Agro)-Institut national d'enseignement supérieur pour l'agriculture, l'alimentation et l'environnement (Institut Agro) | Cognition, Action, et Plasticité Sensorimotrice [Dijon - U1093] (CAPS) ; Université de Bourgogne (UB)-Institut National de la Santé et de la Recherche Médicale (INSERM) | Centre d'Investigation Clinique 1432 (Dijon) - Module Plurithématique : Périnatalité Cancérologie Handicap et Ophtalmologie (CIC-P803) ; Université de Bourgogne (UB)-Direction Générale de l'Organisation des Soins (DGOS)-Institut National de la Santé et de la Recherche Médicale (INSERM)
Review. Correspondence to: [email protected]. Supplementary digital material pp. 14-46
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Показать больше [+] Меньше [-]Английский. INTRODUCTION: Neuromuscular diseases (NMDs) include a large group of heterogeneous diseases. NMDs frequently involve gait disorders, which affect quality of life. Several walking tests and tools have been described in the literature, but there is no consensus regarding the use of walking tests and tools in NMDs or of their measurement properties for walking outcomes. The aim of this review is to present an overview of walking tests, including their measurement properties when used in adults with inherited or genetic NMDs. The aim is to help clinicians and researchers choose the most appropriate test for their objective.EVIDENCE ACQUISITION: A systematic review was conducted after consulting MEDLINE (via PubMed), EMBASE, Science direct, Google Scholar and Cochrane Central Register of Controlled Trials databases for published studies in which walking outcome measurement properties were assessed. The validity, reliability, measurement error and responsiveness properties were evaluated in terms of statistical methods and methodological design qualities using the COnsensus-based Standards for the selection of health Measurement Instruments (COSMIN) guidelines.EVIDENCE SYNTHESIS: We included 46 studies in NMDs. These studies included 15 different walking tests and a wide variety of walking outcomes, assessed with six types of walking tools. Overall, the 6MWT was the most studied test in terms of measurement properties. The methodological design and statistical methods of most studies evaluating construct validity, reliability and measurement error were “very good.” The majority of outcome measurements were valid and reliable. However, studies on responsiveness as minimal important difference or minimal important change were lacking or were found to have inadequate methodological and statistical methods according to the COSMIN guidelines.CONCLUSIONS: Most walking outcomes were found to be valid and reliable in NMDs. However, in view of the growing number of clinical trials, further studies are needed to clarify additional measurement properties.
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