Background: Antegrade percutaneous ureterolithotripsy (URSL) could be a treatment option for large and/or impacted proximal ureteral stones, which are difficult to treat. To review the current approach and treatment outcomes and to compare the efficacy of retrograde and antegrade URSL for large proximal ureteral stones, we evaluated the unique perspectives of both surgical modalities.Methods: This systematic literature review and meta-analysis was performed in July 2020. Articles on human studies and treatment of ureteral stones with URSL were extracted from the PubMed, MEDLINE, Embase, Cochrane Library, Scopus, and the Japan Medical Abstracts Society databases without any language restrictions. The risks of bias for randomized controlled trials (RCTs) and non-randomized controlled trials (non-RCTs) were assessed using the Cochrane risk of tool and the Risk of Bias in Non-randomized Studiesof Interventions tool, respectively.Results: A total of 10 studies, including seven RCTs and three non-RCTs, were selected for the analysis; 433 and 420 cases underwent retrograde and antegrade URSL, respectively. The stone-free rate (SFR) was significantly higher in antegrade URSL than in retrograde URSL (SFR ratio: 1.17, 95% CI: 1.12-1.22; P<0.001), while the hospital stay was significantly longer in antegrade URSL than in retrograde URSL (standardized mean difference: 2.56, 95% CI: 0.67-4.46; P=0.008). There were no significant differences in the operation time and the overall complication rate between the two approaches.Conclusions: Despite the heterogeneity of data and bias limitations, this latest evidence reflects real practice data, which may be useful for decision making.