A 25-year-old man was admitted to our medical center after experiencing prolonged weakness and splenomegaly. A complete blood cell analysis indicated a white blood cell count of 81,190/μL, a platelet count of 206,000/μL, and a hemoglobin concentration of 10.5 g/dL. Peripheral blood analysis showed all stages of neutrophilic maturation, with 1% myeloblasts, 4% promyelocytes, 10% myelocytes, 17% metamyelocytes, 2% eosinophils and 9% basophils. Bone marrow aspirates revealed that the patient was in the chronic phase of chronic myeloid leukemia (CML), with 2.5% myeloblasts, 5.5% promyelocytes, 13% myelocytes, 13% metamyelocytes, 1.5% eosinophils and 9% basophils. A multiplex PCR screen for gene fusions typical of leukemia was negative. Chromosome analysis of the bone marrow showed the presence of 46,XY,t (5;17) (q32;q11) []. Subsequently, dual-color fluorescent in situ hybridization (FISH) proved positive for PDGFRB gene rearrangement. After one week of imatinib treatment at 400 mg/day, the patient achieved CHR. This dosage was then decreased to 200 mg/day, and the patient acquired CCyR three months later. Thereafter, the patient took 100 mg/day, and at the final follow-up (26 months later), the patient still remained in complete remission.