Background: Delusional misidentification syndromes are conditions in which the patients pathologically misidentify people, places, objects or events. They have been categorized in four subtypes: Capgras, Frégoli, intermetamorphosis and subjective double syndromes. Such syndromes may be present in patients with psychiatric disorders such as schizophrenia and mood disorders, and with neurological diseases such as Alzheimer, Parkinson and brain injury (trauma, vascular). Objectives: To describe and discuss a case of coexistent between Capgras and Frégoli syndromes in a female patient with paranoid schizophrenia and brain MRI findings. Methods: Psychiatric interview and brain MRI scanning. Results: The patient presented structural magnetic resonance imaging periventricular and subcortical white matter hyperintensities on flair images mainly concentrated in the right frontotemporal region and bilateral frontotemporal volume loss. Discussion: The described neuroimaging findings may represent an organic substrate to the delusional misidentification syndromes of the present case. The delusional symptoms in Capgras and Frégoli syndromes could be the result of a right temporolimbic-frontal disconnection which results in impossibility to associate previous memories to new information and consequently misidentifying symptoms. Moreover a volume loss of such cerebral regions, as observed in the present case, may also play a significant role in the development of delusional misidentification syndromes.