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We report a case of chronic myelogenous leukemia (CML) in which the bone marrow achieved cytogenetic complete remission (CCR) achieved by treatment with interferon-alpha and oral cytarabine ocfosfate after extramedullary blast crisis. A 51-year-old Japanese man diagnosed with CML was treated with interferon-alpha. Two months later; lymph node swellings developed in his neck and inguinal regions. Lymph node biopsy revealed the infiltration of blast cells showing bcr-abl fusion signal by fluorescence in situ hybridization analysis. Bone marrow aspiration and cytogenetic analysis demonstrated that his bone marrow was still in the chronic phase, with minor cytogenetic response. Continuing interferon-alpha for 6 months in combination with oral cytarabine ocfosfate resulted in the disappearance of the neck lymph node swellings followed by CCR in the bone marrow. However, rapid reenlargement of the neck and inguinal lymph nodes was noted 2 months after CCR despite maintaining medullary remission with major cytogenetic response. Finally, medullary crisis was noted 13 months from the initial development of the extramedullary crisis. This case suggests that interferon-alpha plus cytarabine ocfosfate therapy may be of benefit in the treatment of extramedullary blast crisis of CML.