The central aim of our laboratory is to elucidate the biological roles of RNA modification enzymes. Through the development of new model systems to study RNA modification, our lab has uncovered novel targets and functions for RNA modification enzymes, including the discovery of unanticipated roles for tRNA modification. Notably, our studies have uncovered the molecular basis for human diseases associated with tRNA modification enzymes. Our present studies are aimed at understanding the biological functions of RNA modification enzymes linked to neurodevelopmental and neurological disorders.