We report a 74-year-old lady with hemichorea of 6 months duration due to severe carotid stenosis without cerebral infarction. Electroencephalography and single-photon emission computed tomography studies suggested hemispheric abnormality. Hemichorea resolved following carotid endarterectomy. Hemichorea is the commonest movement disorder following acute stroke. It is rarely reported in association with severe carotid stenosis without evident infarction. In the absence of corresponding structural abnormalities on magnetic resonance imaging in hemichorea, vascular imaging is critical in establishing the diagnosis. Hemichorea in setting of carotid or vascular stenosis is often resistant to medical treatment and best responds to reperfusion. Hemichorea due to critical hemispheric ischemia is a well-described rare entity that best responds to reperfusion strategies. Awareness is essential to identify this entity and manage appropriately.

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