Cerebral hyperperfusion syndrome is a rare but severe complication of carotid artery stenting or carotid endarterectomy. Staged angioplasty is reportedly an effective strategy to avoid cerebral hyperperfusion syndrome. We encountered a case of internal carotid artery stenosis with a rare clinical presentation of limb shaking that was successfully improved by staged angioplasty. To our knowledge, there are no reported cases of limb shaking treated with staged angioplasty. A 76-year-old woman presented with continuous chorea in her left lower limb and shoulder. Medical examination revealed a tiny cerebral infarction in the right corona radiata and severe right internal carotid artery stenosis. Angiography showed near occlusion of the right internal carotid artery. Staged angioplasty was performed to avoid the risk of cerebral hyperperfusion syndrome. The first angioplasty resulted in an expanded diameter of 2.5 mm and was followed by definitive carotid artery stenting using a closed-cell stent 3.5 weeks later. Limb shaking improved in a stepwise manner along with an improvement in internal carotid artery stenosis and distal flow state with no signs of cerebral hyperperfusion syndrome. Patients with internal carotid artery stenosis or occlusion presenting with limb shaking have been suggested to have impaired cerebrovascular reactivity, which is also thought to be a risk factor for cerebral hyperperfusion syndrome. The stepwise improvement in limb shaking observed in this case supports the idea that the pathophysiology of limb shaking is related to cerebral haemodynamic impairment. Measures to prevent cerebral hyperperfusion syndrome, including staged angioplasty, should be actively considered in patients with limb shaking because the symptoms themselves suggest severe hypoperfusion.
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