Obsessive–Compulsive Disorder (OCD) is a chronic, severe disabling neuropsychiatric disorder whose pathophysiology is not yet well defined. Generally, the symptom onset occurs during pre-adult life and affects subjects in different life aspects, including professional and social relationships. Although robust evidence indicates the presence of genetic factors in the etiopathology of OCD, the entirely mechanisms are not totally clarified. Thus, the possible interactions between genes and environmental risk factors mediated by epigenetic mechanisms should be sought. Therefore, we provide a review of genetic and epigenetic mechanisms related to OCD with a deep focus on the regulation of critical genes of the central nervous system seeking possible potential biomarkers.