Olfactory reference syndrome (ORS) is a rare psychiatric condition with an unclear etiology showing great interindividual variance in its course and treatment response. Although the core symptom of the disease is preoccupation with imagined body odor that persists despite reassurance, clinical presentations mimic various psychiatric illnesses. There have been several case reports of underlying general medical conditions related to ORS, such as epilepsy, arteriovenous malformation and substance abuse. However, to our knowledge, there has been no report on regional blood flow changes detected in patients with ORS. In this brief report, we present the case of a patient with ORS who underwent unnecessary surgery and was referred twice by surgeons. The phenomenology, nosological features and differential diagnosis of ORS are also discussed with regard to its relationship with affective disorders.