Dysregulation of microRNA networks has been implicated in hematological malignancies. One of the reasons for disturbed miRNA-mediated regulation are polymorphisms in miRNA-binding sites (miRSNPs), which alter the strength of miRNA interaction with target transcripts. In the recent years the first findings of miRSNPs associated with risk and prognosis in hematological malignancies have been reported. From the studies described in this review miRSNPs not only emerge as novel markers of risk and prognosis but can also lead to better understanding of the role of miRNAs in regulating gene expression in health and disease.