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Easily missed or misinterpreted: diabetic striatopathy in the course of ketotic hyperglycaemia

© Author(s) (or their employer(s)) 2021. No commercial reuse. See rights and permissions. Published by BMJ. A 61yearold man suffering from diabetes mellitus type 2 (DM2) for 10 years and a 67yearold woman with no medical history developed subacutely unilateral involuntary movements. The former patient demonstrated moderate hemichoreahemiballism (HCHB) in the left limbs for 3 weeks, while the latter patient presented mild hemichorea in the right upper limb for 1 week (online supplemental video showing symptoms of the second patient). Laboratory tests revealed hyperglycaemia (random blood glucose levels: 470 mg/dL and 560 mg/dL, respectively) and elevated glycosylated haemoglobin A1c levels (15.4% and 13.1%, respectively) with positive urine ketones and normal blood gas analysis. Both brain CT and MRI showed lesions in the striatum contralateral to the clinical symptoms and similar to those described in a nonketotic hyperglycaemia with HCHB (figures 1A,B and 2A,B). Insulin glucoselowering treatment led to the normalisation of glycaemia and urine ketones within a few days, and complete resolution of involuntary movements. At a 6month followup visit, both patients remained asymptomatic, despite the persistence of striatal abnormalities in neuroimaging (figures 1C,D and 2C,D). HCHB is a syndrome mostly associated with a nonketotic hyperglycaemia, often described