USP46 belongs to the peptidase C19 family, USP12/USP46 subfamily. Deubiquitinating enzymes (DUBs) are a large group of proteases which are also commonly referred to as deubiquitinating peptidases, deubiquitinating isopeptidases, deubiquitinases, ubiquitin proteases, ubiquitin hydrolyases, ubiquitin isopeptidases, or Dubs. They regulate ubiquitin-dependent metabolic pathways by cleaving ubiquitin-protein bonds. They also may act as negative and positive regulators of the ubiquitin system. Besides ubiquitin recycling, they are also involved in processing of ubiquitin precursors, in proofreading of protein ubiquitination and in disassembly of inhibitory ubiquitin chains. USP46 is a deubiquitinating enzyme that plays a role in behavior, possibly by regulating GABA action. It may act by mediating the deubiquitination of GAD1/GAD67. USP46 has almost no deubiquitinating activity by itself and requires the interaction with WDR48 to have a high activity and it is not involved in deubiquitination of monoubiquitinated FANCD2.