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Understanding how psychopathy compares with brain disease can help clarify its underlying mechanisms. This literature review is a broad overview of the neurobiology of psychopathic traits in comparison to behavioral variant frontotemporal dementia (bvFTD), a disorder uniquely associated with criminal behavior. In addition to violation of social norms, both psychopathy and bvFTD result in impaired socioemotional perception and empathy, impulsivity, and altered moral judgment. Despite wide areas of decreased function in psychopathy, structural changes are primarily evident in amygdala and, to a lesser extent, anterior insula, whereas in bvFTD neuropathology involves a wider paralimbic region. In psychopathy, relatively intact medial prefrontal and anterior cingulate cortices facilitate theory of mind and psychopathic traits such as deceitfulness and manipulation, bold fearlessness, and risk-taking behavior. In conclusion, many frontotemporal areas are hypoactive in psychopathy and bvFTD, but differences in dysfunctional connectivity in psychopathy vs. direct involvement in bvFTD potentially explain similarities and differences between these two conditions.