Levosulpiride-Associated Hemichorea

To the Editor: Hemichorea is usually associated with a unilateral lesion of the central nervous system. Causative brain lesions include stroke, neoplasm, vascular malformation, and tuberculoma. 1 Drugs and systemic illness have been linked to hemichorea without brain lesions; however, such instances are extremely uncommon. 2-5 Levosulpiride is a highly selective dopamine D2-receptor antagonist that is widely used for the treatment of functional dyspepsia. It is reported to induce extrapyramidal syndromes , 6 although there is no description of hemichorea. A 63-year-old, right-handed woman with a history of hyper-lipidemia presented at our clinic with increasing involuntary, irregular, and purposeless movements of her lip, left arm, and leg (Supplementary Video 1, only online). Ten days prior, her daughter noticed these abnormal movements. The patient denied any family history of movement disorders. The movements were enhanced with action (i.e., walking) and disappeared during sleep. There were no fluctuations during the day. Neurological examination was normal except for the involuntary movements. Brain MRI, including diffusion-weighted imaging (DWI), was normal except for a benign-looking vas-cular malformation (i.e., cavernous angioma) in the left medial temporal area, and MR angiography produced normal findings (Fig. 1A and B). Her laboratory findings were unremark-able, including fasting glucose, glycated hemoglobin A, glucose tolerance, thyroid function, venereal disease research laboratory , peripheral blood smear, creatine kinase, antinuclear an-tibody, anticardiolipin antibody, anti-neutrophil cytoplasmic antibodies, antiphospholipid antibody, rheumatoid factor, and antistreptolysin O. Routine urinalysis and 24-hour urine collection (for creatinine clearance and protein excretion evaluation) results showed normal renal function. Somatosenso-ry-evoked potentials for median and posterior tibial nerve stimulation were normal. The patient had been taking levo-sulpiride (75 mg/day), talniflumate (1110 mg/day), ranitidine (300 mg/day), afloqualone (60 mg/day), and calcium polycar-bophil (1875 mg/day) for about 2 months for her arthralgia and gastrointestinal discomfort. Five days after complete medication withdrawal, the involuntary movements significantly subsided. Single photon emission computed tomography (SPECT), using 99m technetium-hexylmethylpropylene amine oxime (99m Tc-HMPAO) at day 5, showed mild hypoperfusion in the right basal ganglia, particularly the caudate nucleus (Fig. 1C). Although her involuntary movements were improving , she continued to complain of the involuntary movements, and quetiapine (12.5 mg) was prescribed nightly. We could not increase the dosage and were forced to stop the medication after 1 week, as she complained of drowsiness. We could not determine the effect of quetiapine due to the small dose. Within 1 month, the hemichorea completely resolved. During the 11-month follow-up period, …