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Isolated chloromas(granulocytic sarcomas) are rare tumors. Chloromas are masses composed of immature granulocytic cells. Granulocytic sarcoma occurs primarily in patients with acute myelogenous leukemia as well as in patients with other myeloproliferative disorders, but rarely in patients with acute lymphoblastic leukemia(ALL). We now describe one patient affected by ALL with isolated granulocytic sarcoma as initial CNS relapses. These unusual clinical manifestation and radiological finding in acute lymphoblastic leukemia should be considered as recurrence of leukemia. Early detection and antileukemic treatment of granulocytic sarcoma are necessarily important for favorable prognosis. Key words:Acute lymphoblastic leukemia;Chloroma;Granulocytic sarcoma.