The ever-expanding field of epitranscriptomics and RNA post-transcriptional modifications is delving into the significant role RNA modifications play in disease management. The therapeutic application of RNA modifications is emerging as a highly promising avenue for drug development, given the existence of over 170 distinct types of modifications across various RNA species. Critical biological processes implicated in numerous diseases have been found to be regulated by genes associated with these RNA modifications. Current research has identified inhibitors targeting genes that regulate RNA modifications, presenting a compelling proposition for RNA modification as a viable therapeutic target in disease treatment. This chapter aims to delve into the mechanisms by which molecular alterations target RNA modifications or their regulatory genes. It will also consolidate existing research on the impact of RNA modifications and regulatory genes within the sphere of drug development, highlighting the significant potential of targeting RNA modifications in the creation of innovative therapeutic strategies.