Background: Body dysmorphic disorder (BDD) is a psychological state in which patients recognize a distorted perception of their body images. Herein, we aimed to evaluate the prevalence of BDD in patients seeking orthodontic treatment via a systematic review of the scientific literature. Further, in the meta-analysis, the emphasis was to explore the central tendency and level of significance in cases of BDD seeking orthodontic treatment. Data sources: A detailed scientific literature search was conducted utilizing the most relevant scientific studies between 2000 and 2020. We searched various databases, including PubMed, Cochrane Central Register of Controlled Trials, and Scielo databases. The MedCalc tool was used for the meta-analysis. Data selection: We analyzed a total of 1745 participants enrolled in five different studies. The clinical diagnosis results showed 122 cases of BDD-YBOCS (based on the Yale-Brown Obsessive- Compulsive Scale for BDD score) seeking orthodontic treatment. A significant increase (5–10%) in the number of BDD cases opting for orthodontic treatment was noted, and female patients with BDD showed a higher tendency of seeking orthodontic treatment than the male patients. Considering the forest plot and funnel plot analyses, a significant increase in the cases of BDD was observed (proportions; P<0.0001). Conclusions: The patients with BDD seeking orthodontic treatment have increased over the last two decades (range: 5–10%). Orthodontists are recommended for these cases of BDD seeking multiple consultations.