Purpose: To present a case of acute B-cell lymphoblastic leukemia (B-ALL) ocular relapse treated with chimeric antigen receptor T-cell (CAR-T) therapy. Methods: A retrospective case review was evaluated. Results: A 69-year-old woman with a history of B-ALL presented with bilateral hypopyons and 3+ anterior chamber cells that responded to topical prednisolone. Six months later, she developed recurrent hypopyons, iris neovascularization, and iris nodularity in both eyes. A fine-needle aspiration biopsy confirmed ocular infiltration of B-ALL. A single infusion of CAR-T therapy resulted in remission of the systemic and ocular findings. The clinical course was complicated by cytokine release syndrome and neurotoxicity, which improved with medical management. The patient remained in remission for 7 months after a single CAR-T infusion. Conclusions: CAR-T therapy may be effective in treating systemic leukemia and uveal infiltration, with a favorable ocular side-effect profile and visual outcomes. The systemic side effects of CAR-T therapy may be managed medically.
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