Bilateral dystonia in type 1 diabetes: a case report

A 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.Hyperglycemia-induced involuntary movement is one of the manifestations of dystonia and hemichorea-hemiballism.Chorea is defined as irregular, unpredictable, brief and jerky involuntary movements, while ballismus is large-amplitude flailing movements [1]. Hemichorea-hemiballismus is a rare complication of non-ketotic hyperglycemia and only 53 case reports of this particular condition were published between 1985 and 2001 [2]. Most of the cases were over 60 years of age and represented type 2 diabetes and non-ketotic hyperglycemia. The differential diagnosis of diabetic hemichorea-hemiballismus is challenging because this type of hyperkinetic movement disorder is caused by focal lesions, such as ischemic or hemorrhagic stroke, infection, epilepsy, and neoplasm, as well as systemic processes, including systemic lupus erythematosus, Wilson's disease and thyrotoxicosis [1]. Here, we present a case with type 1 diabetes dystonia manifesting ashyperglycemia-induced involuntary movement.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 also presented with hemichorea and bilateral dystonia for 5 days. She forefelt onset for several seconds before initiati