BackgroundCatatonia is a complex psychomotor disorder characterized by motor, affective, and behavioral symptoms. Despite being known for almost 150 years, its pathomechanisms are still largely unknown. MethodsA systematic research on PubMed, Web of Science, and Scopus was conducted to identify neuroimaging studies conducted on group or single individuals with catatonia. Overall, 33 studies employing structural magnetic resonance imaging (sMRI, n = 11), functional magnetic resonance imaging (fMRI, n = 10), sMRI and fMRI (n = 2), functional near-infrared spectroscopy (fNIRS, n = 1), single positron emission computer tomography (SPECT, n = 4), positron emission tomography (PET, n = 4), and magnetic resonance spectroscopy (MRS, n = 1), and 171 case reports were retrieved. ResultsObservational sMRI studies showed numerous brain changes in catatonia, including diffuse atrophy and signal hyperintensities, while case-control studies reported alterations in fronto-parietal and limbic regions, the thalamus, and the striatum. Task-based and resting-state fMRI studies found abnormalities located primarily in the orbitofrontal, medial prefrontal, motor cortices, cerebellum, and brainstem. Lastly, metabolic and perfusion changes were observed in the basal ganglia, prefrontal, and motor areas. Most of the case-report studies described widespread white matter lesions and frontal, temporal, or basal ganglia hypoperfusion. ConclusionsCatatonia is characterized by structural, functional, perfusion, and metabolic cortico-subcortical abnormalities. However, the majority of studies and case reports included in this systematic review are affected by considerable heterogeneity, both in terms of populations and neuroimaging techniques, which calls for a cautious interpretation. Further elucidation, through future neuroimaging research, could have great potential to improve the description of the neural motor and psychomotor mechanisms underlying catatonia.