BACKGROUND:Aquatic therapy has several proposed benefits for people with lymphedema. A systematic review of the evidence for aquatic therapy in lymphedema management has not been conducted. METHOD AND RESULTS:Systematic review and meta-analysis were conducted. Five electronic databases were searched to identify randomized controlled trials (RCTs) of people with lymphedema, which compared aquatic therapy with other lymphedema interventions. Qualitative analysis was undertaken where quantitative analysis was not possible. Study quality was assessed using physiotherapy evidence database (PEDro) scores. The strength of evidence was evaluated using the Grades of Recommendations Assessment, Development and Evaluation (GRADE) approach. Four RCTs of moderate quality (average PEDro score 6.5/10) were included in the review. Two studies provided results for inclusion in meta-analysis. There was moderate-level evidence of no significant short-term differences in lymphedema status (as measured by lymphedema relative volume) between patients who completed aqua lymphatic therapy (ALT) compared to land-based standard care (standardized mean difference [SMD]: 0.14; 95% confidence interval [CI]: -0.37 to 0.64, I2 = 0%, p = 0.59); and low-quality evidence of no significant difference between ALT and standard care for improving upper limb (UL) physical function (SMD -0.27, 95% CI: -0.78 to 0.23, I2 = 0%, p = 0.29). No adverse events reported. CONCLUSIONS:Current evidence indicates no significant benefit of ALT over standard land-based care for improving lymphedema status or physical function in people with UL lymphedema. Patient preference should guide the choice of care to facilitate adherence. Further research is required to strengthen the evidence from four studies in people with UL lymphedema, and to establish the efficacy of this intervention in people with lower limb lymphedema. Review registration: PROSPERO (CRD42015019900).