The authors report follow-up observations for a case of isolated leukemic uveopathy that was first diagnosed in an 11-year-old girl shortly after cessation of treatment for acute lymphoblastic leukemia (ALL). Prior therapy for the complication included systemic antiblastic chemotherapy and low doses of radiation (3.9 Gy) to the affected eye. In July 1979, 2 months after chemotherapy was stopped for the second time, she presented with recurrent leukemic hypopyon in the left eye and was treated again with antiblastic chemotherapy followed in 1 year by high-dose (20 Gy) local irradiation. After a third recurrence, which was diagnosed as pre-B/B-cell ALL with a variable position of maturation arrest, enucleation was performed. The patient remains in complete remission for 26+ months after an additional course of systemic chemotherapy. This case illustrates the difficulty of eradicating leukemic cells from the eye with conventional treatment but suggests that a relapse in this site is not necessarily an ominous prognostic sign.