We report on a patient with chronic myeloid leukemia (CML) with a rapidly growing left cervical tumor 5 months after the initial diagnosis of CML. This tumor was diagnosed as a very early manifestation of extramedullary myeloblastoma by a minimally invasive method. A fine needle aspirate (26-gauge needle) was obtained from the tumor. Morphological and cytochemical analysis of the aspirate revealed 19% undifferentiated blasts. The immunophenotype was suggestive of a myeloid differentiation of the blasts (CD33+). The CML origin of the blasts was confirmed by the detection of the bcr-abl gene rearrangement in the blasts by two-color fluorescence in situ hybridization (FISH). We conclude that fine needle aspiration in combination with immunophenotyping and FISH analysis of the aspirate is a minimally invasive and rapid diagnostic tool to confirm extramedullary manifestation in CML. To our knowledge, this is the first case of extramedullary myeloblastoma confirmed by this combined technique.