Leukemia showing relapse in the eye, may have the ocular manifestations ranging from leukemic retinopathy to involvement of the iris and anterior chamber. Ocular manifestations presenting as hypopyon may occur in relapse Acute lymphoblastic leukemia (ALL), Acute Myeloid Leukemia (AML), and Chronic Myeloid Leukemia (CML). We report a case of unilateral hypopyon as a rare presenting feature of relapsed Acute Lymphoblastic Leukemia (ALL) in a paediatric patient. Relapse in ALL usually occurs in bone marrow, CSF and testis. Ocular manifestations are extremely rare. We report a 5- yearold male child presented with unilateral hypopyon after completion of chemotherapy. Anterior chamber aspirate showed malignant cells (leukemic infiltrates). Subsequently, he was found to have relapse in bone marrow and CSF too. After initiation of chemotherapy, his hypopyon resolved and he is doing well in follow up.