An 81-year-old woman with poorly controlled diabetes mellitus was hospitalized due to hemichoreahemiballism. A radiological examination revealed typical putaminal changes of diabetic hemichoreahemiballism (DHC-HB). Interestingly, brain computed tomography, performed before symptom onset, disclosed a hyperdense lesion in the left basal ganglia, indicating persistent basal ganglia impairment, even before the onset of symptoms, under sustained hyperglycemia. Additionally, an increase in the cerebrospinal fluid level of homovanillic acid was related to the symptom appearance of DHC-HB. Pronounced potential basal ganglia impairment under hyperglycemia and central dopaminergic hyperactivity was important for the development of DHC-HB in this patient.