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Objective: To evaluate the effects and safety of exercise training, and to determine the most effective exercise intervention for people with Duchenne muscular dystrophy. Exercise training was compared with no training, placebo or alternative exercise training. Primary outcomes were functioning and health-related quality of life. Secondary outcomes were muscular strength, endurance and lung function. Data sources: A systematic literature search was conducted in Medline, EMBASE, CINAHL, Cochrane Central, PEDro and Scopus. Study selection and data extraction: Screening, data extraction, risk of bias and quality assessment were carried out. Risk of bias was assessed using the Cochrane Collaborations risk of bias tools. The certainty of evidence was assessed using Grading of Recommendations Assessment, Development and Evaluation. Data synthesis: Twelve studies with 282 participants were included. A narrative synthesis showed limited or no improvements in functioning compared with controls. Health-related quality of life was assessed in only 1 study. A meta-analysis showed a significant difference in muscular strength and endurance in favour of exercise training compared with no training and placebo. However, the certainty of evidence was very low. Conclusion: Exercise training may be beneficial in Duchenne muscular dystrophy, but the evidence remains uncertain. Further research is needed on exercise training to promote functioning and health-related quality of life in Duchenne muscular dystrophy. Lay abstract The aim of this study was to examine the effects of all types of exercise training compared with no training, placebo or alternative exercise training programmes in people with Duchenne muscular dystrophy. The primary outcomes were functioning and health-related quality of life. Secondary outcomes were muscular strength and endurance. A further aim was to evaluate safety and, if possible, to find the most effective exercise training intervention. This review investigates existing research on this topic. The results have been systematically gathered and analysed, to give a broader view of what effect exercise training may have for people with Duchenne muscular dystrophy. The results suggest that exercise training preserves functioning, and benefits muscular strength and endurance. The study was not able to identify the best type of exercise training and prescriptions to use in Duchenne muscular dystrophy. The validity of the results was reduced by the low number of studies included, the low quality of the studies, and diversity in both the interventions and outcome measures used. The results should therefore be interpreted with caution.