Between 1988 and 1992, seven patients with overt meningeal leukemia who had received adequate central nervous system (CNS) prophylaxis were treated with intermittent craniospinal irradiation and intrathecal methotrexate(IIIC). Follow-up time ranged from 8 months to 41 months with median of 20 months. Three of 7 patients developed subsequent CNS relapse. CNS remission durations were 8, 9, 13, 20, 28, 34, 36 months from diagnosis of CNS leukemia for which IIIC was given. Disease free survival after CNS relapse ranged from 2 to 36 months with median of 11 months. Overall survival after CNS relapse ranged from 8 to 41 months with median of 28 months. Five patients died of sepsis and bleeding secondary to bone marrow relapse. Two patients are alive at present. But they developed recurrent CNS disease 10 to 11 months after completion of IIIC. To improve the outcome, modification of IIIC by reduction of rest period and prolonged administration of intrathecal chemotherapy after completion of IIIC are required.