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Isolated extramedullary ocular relapse of acute lymphoblastic leukemia (ALL) after allogeneic peripheral blood stem cell transplantation (allo-PBSCT) without concomitant involvement of the bone marrow is very rare,while the common sites of extramedullary relapse are the central nervous system,skin,bone,and breasts.1 This is the report of isolated ocular relapse without any extraocular involvement of ALL after allo-PBSCT confirmed by histopathology.