General prevalence of Body Dysmorphic Disorder (BDD), a psychiatric disorder in which patients focus on an imagined body defect not visible to others, varies between 0.7 and 2.5%. Up to 86% present with complaints in the area of teeth or face. Patients with BDD pursue an aesthetic rather than functional recovery, making BDD a possible contraindication to treatment. The aim of this systematic review was to review prevalence of BDD within the orthodontic and/or orthognathic population, to describe diagnostic tools for early detection of patients with BDD and to assess the outcome after treatment. A systematic search was conducted up to November 2020 using PubMed, Embase, Web of Science Core Collection and Cochrane Library. Following the screening of 1423 articles, 5 prospective studies were included. Prevalence of BDD within the orthodontic and orthognathic population varied from 5.2% to 13% (average of 6.2%). Literature showed a trend of higher BDD prevalence within younger, female and single patient population, although results are not conclusive. Questionnaires can be useful for preliminary detection of BDD. General anamnesis with questioning medical or psychiatric history, medication and personal expectations remains very important. Red flags could be previous consultations for the same problem or presence of psychiatric comorbidities. Prospective studies are necessary to map satisfaction of these patients and need for re-interventions after treatment.