A 48-year-old woman presented with a 12-month history of recurrent episodes of flushing of her right face after exercise [Figure 1]. During every episode, the flushing resolved after rest. Neurological and ophthalmological examinations were normal. Routine laboratory studies, high-resolution computed tomography of the chest, carotid artery ultrasonography, and magnetic resonance imaging of head, neck, and cervicobrachial plexus were normal. A diagnosis of idiopathic Harlequin syndrome was made. The patient did not receive treatment since she was not bothered by her symptoms and followed up in the clinic with persistence of symptoms and normal health status.Figure 1: Flushing of unilateral faceHarlequin syndrome is characterized by unilateral facial flushing and sweating induced by exercise, heat, and emotion.[1] The syndrome is most common in women, and social embarrassment is the main complaint. Most of Harlequin syndrome are idiopathic in nature. However, Harlequin syndrome could be caused by an underlying disease or iatrogenic injure, such as brainstem infarction, thyroid diseases, avulsion injury of brachial plexus, pancoast tumor, thoracic surgery, superior mediastinum neurinoma, dissection of internal carotid artery, internal jugular vein catheterization, regional infection, and so on.[2] The treatment depends on the etiology and the patient's need. Most patients do not require medical or surgical treatment unless there is an underlying disease. Declaration of patient consent The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed. Financial support and sponsorship None. Conflicts of interest There are no conflicts of interest.