OBJECTIVE: The aim of this systematic review and meta-analysis was to assess the effect of aerobic, resistance and combined exercise training in patients with ALS. DATA SOURCE: A comprehensive systematic search of CENTRAL, CINAHL, SPORTDiscus, Embase, Scopus, ProQuest was performed from inception to February 2021. ELIGIBILITY CRITERIA: The systematic review included all studies that examined the effect of exercise training in ALS patients. Meta-analysis was also carried out on randomized controlled trials (RCTs). DATA EXTRACTION AND DATA SYNTHESIS: Data related to primary outcomes (functional ability, respiratory function, fatigue, pain, quality of life, upper-body strength, lower-body strength and VO2peak) and secondary outcomes (adverse events and feasibility of exercises) was extracted from all studies and systematically reviewed. RESULTS: 16 trials including 532 patients met the inclusion criteria; of these, eight studies were included in this meta-analysis. The meta-analysis found a statistically significant difference in favor of exercise in functional ability (p = 0.001), overall quality of life (p = 0.03) and VO2peak (p = 0.01). The included trials were generally of poor quality and had a risk of bias. However, the results of sensitivity analysis, after omitting studies with high risk of bias, showed no statistically significant difference in functional ability (p = 0.05), overall quality of life (p = 0.12) and VO2peak (p = 0.13). Finally, no significant difference was found in respiratory function, fatigue, pain, and upper-body and lower-body strength. CONCLUSIONS: The safety and effectiveness of exercise therapy in ALS patients remains unclear and further high quality RCTs with larger sample size are needed. SYSTEMATIC REVIEW REGISTRATION NUMBER: PROSPERO CRD42019140011.

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