Several studies reported the beneficial effects of globus pallidus internus deep brain stimulation (GPi DBS) on health-related quality of life (HRQoL) in patients with inherited or idiopathic isolated dystonia. However, the impact of this intervention on physical and mental/psychological domains and the effects over time remain unclear.We conducted a systematic literature review from January 2000 to May 2019 and performed a meta-analysis of HRQoL outcomes based on the Short Form Health Survey-36 (SF-36) after GPi DBS in patients with inherited or idiopathic isolated dystonia to evaluate the effects of DBS on physical and mental QoL.Seven studies comprising 144 patients with dystonia (78, generalised; 34, segmental; and 32, focal cervical) were included in this comprehensive analysis. The mean (SD) age at DBS implantation was 41.0 (11.4) years, and the follow-up period after implantation was 3.2 (3.8) years. The random effects model meta-analysis revealed that both physical and mental domains of SF-36 improved following DBS with a significantly larger effect size for the physical domains (effect size=0.781; p<0.0001) compared with the mental domains (effect size=0.533; p<0.0001). A moderator variable analysis demonstrated that effect sizes for HRQoL improvement were maintained over time.This is the first meta-analysis that demonstrates significant benefits in HRQoL following DBS in patients with inherited or idiopathic isolated dystonia. The benefits are greater for physical QoL domains compared with mental/psychological QoL. These findings highlight the importance of a comprehensive multidisciplinary approach to improve mental/psychological QoL.