Leukemic relapse of the eye has sometimes been cured. In this paper we review the cases of leukemic infiltration of the eye tissue with the aim of ascertaining whether an optimal treatment can be suggested. Data from 25 children (16 males, 9 females) with isolated ocular relapse (10 in therapy, 15 off therapy) of acute lymphoblastic leukemia (ALL) in first complete remission are examined. The patients were treated according to different chemotherapy schedules, with (20 pts) or without local radiotherapy (5 pts). Isolated ocular relapse can be successfully treated, especially if it occurs after the withdrawal of therapy; second ocular infiltration was seen only in anterior chamber involvement after low doses of local radiotherapy. While the need for high doses of chemotherapy is not evident, high doses of ocular radiotherapy (> 20 Gy) seem to be mandatory to cure this leukemic relapse.