Infants with B-cell acute lymphoblastic leukemia (B-ALL) have poor outcomes due to chemotherapy resistance leading to high relapse rates. Tisagenlecleucel, a CD19-directed chimeric antigen receptor T-cell (CART) therapy, is FDA approved for relapsed or refractory (R/R) B-ALL in patients <25 years; however, the safety and efficacy of this therapy in young patients is largely unknown since children <3 years of age were excluded from licensing studies. We retrospectively evaluated data from the Pediatric Real-World CAR Consortium to examine outcomes of patients with infant B-ALL who received tisagenlecleucel between 2017 and 2020 (n=14). Sixty-four percent of patients (n=9) achieved minimal residual disease (MRD)-negative remission post-CART and 50% of patients remain in remission at last follow-up. All patients with high disease burden at time of CART infusion (>M1 marrow) were refractory to this therapy (n=5). Overall, tisagenlecleucel was tolerable in this population, with only 3 patients experiencing ≥ grade 3 cytokine release syndrome. No neurotoxicity was reported. This is the largest report of tisagenlecleucel use in infant B-ALL and shows that this therapy is safe and can be effective in this population. Incorporating this novel immunotherapy into the treatment of infant B-ALL offers a promising therapy for a highly aggressive leukemia.