Hyperthyroidism and moyamoya syndrome (MMS) are rare causes of chorea. 1 Here, we report a patient who had chorea associated with both conditions.A 19-year-old woman from Ivory Coast was admitted because of movement disorder. There was no family history of neurological diseases. The patient had been diagnosed with Graves' disease at age 18 and had since showed poor compliance with carbimazole therapy. Upon admission, free thyroxine level was more than 100 pmol/L (range 12-23), thyroid stimulating hormone was undetectable (less than 0.001 mUI/L, range 0.3-4.4), and thyroid stimulating antibodies were positive: 21.1 UI/L. About 2 weeks before admission, she had progressively developed movement disorder, balance impairment, and dysarthria. On examination, the patient had a diffuse goiter and typical signs of thyrotoxicosis, including warm, moist skin, and tachycardia. She had mild and diffuse choreic movements, predominating in the facial area. Movements involved symmetrically both upper limbs, mainly in sustained posture. The patient had mild dysarthria and tandem walk was impaired. Mild slowing of alternating movements of both hands was present. Neurological examination was otherwise normal. Routine blood count, electrolytes, urea, creatinine, liver function panel, were all normal. Autoantibody screen was negative for anticardiolipids, lupus anticoagulant, Antibeta2GP1, antinuclear antibody, and anti dsDNA antibody. Brain magnetic resonance imaging (MRI) showed high signal intensity lesions on FLAIR and T2 sequences in the left anterior putamen and the head of the left caudate nucleus (Fig. 1). MR angiography showed severe stenosis of the extremity of the internal carotid arteries, at the origin of both middle cerebral arteries, as well as of the left anterior cerebral artery. Four-vessel cerebral angiography showed bilateral severe stenosis of the supraclinoid portion of the internal carotid arteries with MMS (Fig 2.). Treatment with carbimazole (20 mg/day) and levothyroxine (synthetic T4) 50 g/ day was resumed, in association with propanolol. Clinical signs of thyrotoxicosis and neurological symptoms subsided within a few weeks. Three months later, the patient had normal neurological examination and her thyroid tests results were normal. Because of poor observance, two relapses occurred in the following year, consisting of the reappearance of clinical signs of hyperthyroidism and chorea. Thyroidectomy was finally performed and the patient has since remained free of symptoms.The association of hyperthyroidism and chorea has been reported in several patients. 2-6 A causal relationship is likely, as