Dialysis disequilibrium syndrome (DDS) is usually seen in severely uremic patients who are dialyzed aggressively. DDS mostly appeared within 24 hours after hemodialysis (HD) and may last for a few hours. This diagnosis is made by the exclusion of other causes including metabolic and intracranial events and has been recognized for more than 40 years. Few reports described the cerebral radiographic features associated with DDS. We present an 83-year-old uremic patient experiencing DDS at initial HD. DDS-related cerebral radiographic manifestations reported in the literature are reviewed, along with a discussion of the role of neuroimaging in the diagnosis of DDS.