Misophonia is a neuro-behavioral syndrome, phenotypically characterized by heightened arousal of the autonomic nervous system and negative emotional reactivity (eg, irritation, anger, anxiety), in response to a reduction in tolerance to specific sounds. The objectives of this review are to (a) characterize the current state of the misophonia research sector, (b) highlight what can be inferred from the limited scientific literature to inspire the treatment of people with misophonia, and (c) outline a program research on this topic. With this review, we extend previous reviews on this topic by critically examining research investigating the mechanisms of misophonia and the differences between misophonia and other conditions. Furthermore, we integrate this limited but growing literature with basic and applied research from other scientific literatures in an interdisciplinary way.