Hypopituitarism in leukemia is very rare. In addition, central nervous system (cns) relapse and leukemic retino pathy in childhood acute lymphoblastic leukemia (all) have declined with the use of modern systemic chemo therapy that includes cns prophylaxis. Here, we report the case of a 4yearold girl who received chemotherapy and intrathecal therapy without cns radiation after a diagnosis of Bprecursor all without cns involvement. Three months after chemotherapy completion, she presented with lowerextremity weakness and was diagnosed with an isolated cns relapse. Concurrent hypopituitarism and leukemic retinopathy were also found. After receiving craniospinal radiotherapy and systemic chemotherapy, her retinopathy and vision improved. She is now in com plete remission, and she is still on chemotherapy according to the guideline from the Pediatric Oncology Group. Although rare, hypopituitarism and leukemic retinopathy should be taken into consideration in patients with cns involvement by leukemia.