In our analysis, the OS of the entire cohort including patients with CCUS, MDS, MDS/AML and AML was sub-optimal at 26.5 months, with a differential prognostic impact imparted by the two U2AF1 hotspot regions; 37.1 vs 14.2 months (P = 0.008) in patients with high-risk myeloid neoplasm harboring U2AF1S34F and U2AF1Q157PMT, respectively. Here, U2AF1 is linked to acute myeloid leukemia.