Abstract
IntroductionDiabetic hemichorea-hemiballismus is a rare complication of type 2 diabetes. Here, we report a case with type 1 diabetes, with hemichorea and bilateral dystonia manifested as hyperglycemia-induced involuntary movement.Case presentationA 62-year-old Japanese women with body weight loss of 30 kg during the past year developed symptoms of thirst, polydipsia and polyuria. She also presented with hemichorea and bilateral dystonia for 5 days and extremely high plasma glucose (774 mg/dl), hemoglobin A1c (21.2%) and glycated albumin (100%) with ketosis. Based on the presence of glutamic acid decarboxylase antibodies (18,000 U/ml; normal <1.3 U/ml), low daily urinary excretion of C-peptide (7.8 μg), ketosis and human leucocyte antigen typing DR-4, we diagnosed type 1 diabetes mellitus. We treated the patient with a continuous intravenous regular insulin infusion and medication with haloperidol, and dystonia completely disappeared within 3 days.ConclusionHyperglycemia-induced involuntary movement is one of the manifestations of dystonia and hemichorea-hemiballism.
Highlights
Diabetic hemichorea-hemiballismus is a rare complication of type 2 diabetes
Many hypotheses have been reported for the development of diabetic hemichorea-hemiballismus, such as local gamma-aminobutyric acid (GABA) starvation, disinhibition of dopaminergic neurons, local microhemorrhage, microinfarction, demyelination and brain edema [4]
Recent imaging analysis has revealed reduced cerebral glucose metabolism on positron emission tomography (PET) scans with concomitant hyperperfusion in affected basal ganglia seen on single photon emission computed tomography (SPECT) [5]
Summary
Chorea is defined as irregular, unpredictable, brief and jerky involuntary movements, while ballismus is largeamplitude flailing movements [1]. A 62-year-old Japanese women with body weight loss of 30 kg during the past year developed symptoms of thirst, polydipsia and polyuria and was admitted to our hospital She presented with hemichorea and bilateral dystonia for 5 days. She forefelt onset for several seconds before initiation of involuntary movement. Since exactly the same pattern of hemichorea and bilateral dystonic movement occurred intermittently every 10 minutes, she could not stand and had had difficulties in taking meals for 2 days. These movements were observed in both waking and sleep states. After the discontinuation of haloperidol, recurrence of dystonia was not observed
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