This article reports early blastic transformation of chronic myeloid leukemia (CML) in a child following a complete cytogenetic response induced by imatinib mesylate. A 14-year-old Japanese boy was diagnosed with t(9;22) cryptic CML in the chronic phase and treated with imatinib. His response to treatment was slow, but a major cytogenetic response was obtained at 142 days of therapy. However, he developed lymphoid blastic transformation at 9 months. He attained remission with acute lymphoblastic leukemia-type chemotherapy and then successfully received a non-T-cell-depleted allogeneic stem cell transplantation (allo-SCT) with his mother's two loci-mismatched donor cells. A sudden blastic transformation may occur even with a complete cytogenetic response induced by imatinib. CML patients who respond slowly to imatinib may still be candidates for allo-SCT, even when a major cytogenetic response is obtained.