BCR and chronic myelogenous leukemia, BCR-ABL1 positive: Persistent leukocytosis with megalosplenia without any underlying infectious or inflammatory cause is a common manifestation among patients with myeloproliferative neoplasm (MPN), especially chronic myeloid leukemia (CML).[1–3] We report a rare case of myeloid neoplasm with BCR-PDGFRA rearrangement characterized by marked elevation of leukocyte count and megalosplenia, which might improve the focus on this group of diseases and potentially reduce missed diagnoses or misdiagnoses.