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AIM To systematically examine the effectiveness of music therapy on preterm infants in neonatal intensive care unit. BACKGROUND In recent years, the application of music therapy for preterm infants in neonatal intensive care unit has attracted more and more attention because of its clinical effects. However, there still exist disputes among different studies. DESIGN A systematic review and meta-analysis. DATA SOURCES Eleven databases were searched over the period from 1910 -4 November 2019. REVIEW METHODS Papers were selected for analysis in accordance with the PRISMA guidelines. The meta-analysis was carried out by using Review Manager 5.3 software. RESULTS A total of 13 trials involving 1,093 participants were included. Meta-analysis showed music therapy had a significant influence on preterm infant's heart rate, respiratory rate, oral feeding volume, stress level, and maternal anxiety with moderate-to-high heterogeneity among studies. Also, music therapy had no influences on oxygen saturation and behavioural state. CONCLUSIONS Music therapy can not only effectively improve preterm infant's heart rate, stable respiratory rate, and attenuate stress level but also exert positive impact on oral feeding volume. In addition, music therapy also plays a role in reducing maternal anxiety. However, due to the heterogeneity across studies in some outcomes, further studies with larger sample size and more stringent design should be conducted before recommendation. IMPACT Music therapy can significantly improve preterm infant's heart rate, respiratory rate, and stress level, as well as increase oral feeding volume. These results may exert a positive impact on well-being and quality of life in preterm infants in the neonatal intensive care unit. Hospitals can apply music therapy which has been considered a non-pharmacological and no-invasive treatment to preterm infants in the neonatal intensive care unit.