OBJECTIVES: Acute lymphoblastic leukemia (ALL) is an aggressive but potentially curable form of leukemia. Rituximab, an anti-CD20 monoclonal antibody, in addition to standard chemotherapy represents a novel therapeutic option for adults with the Philadelphia chromosome-negative, CD20-positive, B-cell precursor ALL sub-type (CD20+ Ph- BCP-ALL). The objective of this analysis is to determine the economic impact to the Canadian public provincial healthcare payer of rituximab in addition to standard of care (SOC) chemotherapy vs. SOC alone in newly diagnosed CD20+ Ph- BCP-ALL METHODS: A decision analytic model included the following health states over a 15-year time-horizon: event-free survival, relapsed/resistant disease, cure and death. SOC was with the two most widely used chemotherapy regimens in Canada: Hyper-CVAD or Dana Farber Cancer Institute (DFCI). Both regimens contained multiple treatment phases. Event-free survival, overall survival and serious adverse event (SAE) rates were taken from a recent major randomized controlled trial. Costs of the model included: first-, second- and third-line treatment and administration; disease management; palliative care; and SAE-related treatments. Model inputs were sourced from public data, literature and cancer agency input. RESULTS:  Rituximab in addition to SOC resulted in 1.33 greater life-years, 1.16 greater quality-adjusted life-years (QALYs) and $51,679 incremental costs. The resulting mean ICER was $39,563/QALY. At a willingness-to-pay threshold of $100,000/QALY, the probability of being cost-effective was 96%. Decision outcomes were robust to the probabilistic and deterministic sensitivity analyses. CONCLUSIONS: For adults with CD20+ Ph- BCP-ALL, rituximab in addition to SOC was found to be a cost-effective intervention, compared to SOC alone from a Canadian public payer perspective.