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To report the clinical characteristics in a case of extramedullary T-lymphoblastic blast crisis of de novo chronic myelogenous leukemia (CML) so as to improve the understanding of this disease. The clinical characteristics, diagnostic approach and treatment of the patient were retrospectively analyzed, and some related literatures were reviewed. According to resuts of blood routine, bone marrow, chromosome and fusion gene tests, this patient was considered to be CML patient. The cervical lymph node biopsy indicated a T-cell