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Compulsive Sexual Behavior Disorder (CSBD) was recently included in ICD-11 as a new impulse control disorder. While this certainly improved the diagnosis of CSBD, the underlying brain mechanisms of the disorder are still poorly understood. Better description of brain functional deficits is required. Here we investigate patterns of resting-state brain functional connectivity (fc) in a group of CSBD patients compared to a group of healthy controls (HC). A MATLAB toolbox named CONN functional connectivity toolbox was employed to study patterns of brain connectivity. Also correlation between fc and severity of CSBD symptoms and other psychological characteristics, assessed with questionnaires, were examined. We collected resting-state functional magnetic resonance imaging data from 81 heterosexual males: 52 CSBD patients and 29 HC. We found increased fc between left inferior frontal gyrus and right planum temporale and polare, right and left insula, right Supplementary Motor Cortex (SMA), right parietal operculum, and also between left supramarginal gyrus and right planum polare, and between left orbitofrontal cortex and left insula when compared CSBD and HC. The decreased fc was observed between left middle temporal gyrus and bilateral insula and right parietal operculum. No significant correlations between psychological questionnaires assessing CSBD symptoms and resting-state functional connectivity were observed. Results from our study extend the knowledge of brain mechanisms differentiating CSBD from HC. The study was the first large sample study showing 5 distinct functional brain networks differentiating CSBD patients and HC. However, the sample was limited only to heterosexual men, in the future a greater diversity in studied sample and longitudinal studies are needed. Also, the present study examined functional connectivity at the level of regions of interest (ROIs). Future studies could verify these results by examining functional connectivity at the voxel level. The identified functional brain networks differentiate CSBD from HC and provide some support for incentive sensitization as mechanism underlying CSBD symptoms. The correlation between psychological assessment (ie, severity of CSBD, depression and anxiety symptoms, level of impulsivity and compulsivity) and resting-state functional connectivity need further examination. Draps M, Adamus S, Wierzba M, et al. Functional Connectivity in Compulsive Sexual Behavior Disorder - Systematic Review of Literature and Study on Heterosexual Males. J Sex Med 2022;19:1463–1471. [ABSTRACT FROM AUTHOR]