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A systematic review of brain structure and function in body dysmorphic disorder (BDD).The underlying neuropathophysiology of BDD remains largely untested and unknown.We identified the temporal gyrus, limbic system, and prefrontal cortex as important regions in BDD.Abnormalitites within identified regions may contribute to disordered visual perception and emotional arousal in BDD.There is need for future neuroimaging research to test the model discussed.There has been an increase in neuroimaging research in body dysmorphic disorder (BDD), yet little is known about the underlying neurobiological basis of the disorder. We aimed to provide a systematic overview of the literature on the neurobiology of BDD. Two reviewers undertook a search of three electronic research databases: PubMed, PsycINFO, and Google Scholar. The search consisted of synonyms commonly associated with BDD and methods to evaluate brain structure, function, and network organisation. Out of an initial yield of 175 articles, 19 fulfilled inclusion criteria and were reviewed. We identified differences in brain activity, structure, and connectivity in BDD participants in frontostriatal, limbic, and visual system regions when compared to healthy control and other clinical groups. We put forth a neurobiological model of BDD pathophysiology that involves wide-spread disorganisation in neural networks involved in cognitive control and the interpretation of visual and emotional information. This review considers how this model might aid in the development of future research and understanding of BDD.