Nonketotic hyperglycaemia is typically associated with a hemiballism-hemichorea pattern of involuntary movement and is characterized by hyperintense signal within the contralateral striatum on T(1)-weighted magnetic resonance imaging (MRI). An oro-bucco-lingual syndrome and location of the lesions has not been previously described. Herein, we report a patient with oro-bucco-lingual dyskinesia associated with nonketotic hyperglycaemia. MRI revealed characteristic T(1) hyperintense lesions in the bilateral putamen.