Post-stroke movement disorders are uncommon, but comprise an important part of secondary movement disorders. These exert variable and heterogeneous clinical courses according to the stroke lesion and its temporal relationships. Moreover, the predominant stroke symptoms hinder a proper diagnosis in clinical practice. This article describes the etiology, treatment options and prognosis of post-stroke movement disorders.

Movement Disorders Following Cerebrovascular Lesions: Etiology, Treatment Options and Prognosis.

Hemichorea improvement following endarterectomy for internal carotid artery stenosis.

Internal carotid artery (ICA) stenosis has been recently reported to cause hemichorea, mainly in East Asia. The East Asian-specific p.R4810K variant of RNF213, a susceptibility gene for moyamoya disease (MMD), accounts for up to 25% of sporadic ischemic stroke with ICA stenosis cases in East Asia. However, as RNF213-related vasculopathy does not meet the diagnostic criteria for MMD, the creation of a new disease category has been suggested. Here, we report the first case of hemichorea in RNF213-related vasculopathy. An 81-year-old woman was admitted to our hospital with choreic movements in the periphery of the right extremities at rest. Though head magnetic resonance imaging showed no fresh or old cerebral infarction, 123I-iodoamphetamine-single photon emission computed tomography showed cerebral blood flow of < 80% in the anterior territory of the left middle cerebral artery (MCA) in a resting state and cerebrovascular reactivity of < 10% in the broader area supplied by the left MCA after acetazolamide challenge. Head magnetic resonance angiography and digital subtraction angiography revealed left ICA C1 portion stenosis with compromised collateral vessels. Involuntary movements resolved with haloperidol administration within 3 days, without apparent recurrence from continuation of the medication for a year. Genetic testing revealed the presence of the heterozygous RNF213 p.R4810K variant. Chorea is thought to be caused by damage to circuitry connecting the basal ganglia with the cerebral cortex, as found in cases of MMD, which possess aberrant vessels in the basal ganglia. However, aberrant vessels and cerebral infarctions were not observed in the basal ganglia in the current case, decreasing the likelihood of a role in chorea. Alternatively, as RNF213 regulates vascular endothelial function and angiogenesis, dysregulation may impair the neurovascular unit and damage basal ganglia circuitry, contributing to the development of chorea. This case may renew interest in the concept of RNF213-related vasculopathy and the pathophysiological mechanisms behind chorea in ICA stenosis.

/pdf/a-case-of-hemichorea-in-rnf213-related-vasculopathy-s0coul4p8s.pdf

A case of hemichorea in RNF213-related vasculopathy

This is a case of a 71 years old man. Hemichorea appeared in the left half of his body in the middle of November, 2014. Minute hyperintense areas in the white matter near the posterior horn of the right lateral ventricle and in the right parasagittal frontal cortex was shown in MRI diffusion weighted image. Severe stenosis was seen in the right internal carotid artery, and reduction in cerebral blood flow of the right cerebral hemisphere including the basal ganglia was shown in single photon emission computed tomography (SPECT). After having carotid endarterectomy of the right internal carotid artery in January, 2015, hemichorea disappeared, and the cerebral blood flow of the right cerebral hemisphere improved. It is known that hemichorea is caused by the infarction of the basal ganglia. In recent years, some hemichorea cases are reported around East Asia caused by internal carotid artery stenosis with reduction in cerebral blood flow.

A case of hemichorea caused by right internal carotid artery stenosis

Background Studies of secondary movement disorder (MD) caused by cerebrovascular diseases have primarily focused on post-stroke MD. However, MD can also result from cerebral artery stenosis (CAS) without clinical manifestations of stroke. In this study, we aimed to investigate the clinical characteristics of MD associated with CAS. Materials and Methods A nationwide multicenter retrospective analysis was performed based on the data from patients with CAS-associated MDs from 16 MD specialized clinics in South Korea, available between January 1999 and September 2019. CAS was defined as the >50% luminal stenosis of the major cerebral arteries. The association between MD and CAS was determined by MD specialists using pre-defined clinical criteria. The collected clinical information included baseline demographics, features of MD, characteristics of CAS, treatment, and MD outcomes. Statistical analyses were performed to identify factors associated with the MD outcomes. Results The data from a total of 81 patients with CAS-associated MD were analyzed. The mean age of MD onset was 60.5 ± 19.7 years. Chorea was the most common MD (57%), followed by tremor/limb-shaking, myoclonus, and dystonia. Atherosclerosis was the most common etiology of CAS (78%), with the remaining cases attributed to moyamoya disease (MMD). Relative to patients with atherosclerosis, those with MMD developed MD at a younger age (p < 0.001) and had a more chronic mode of onset (p = 0.001) and less acute ischemic lesion (p = 0.021). Eight patients who underwent surgical treatment for CAS showed positive outcomes. Patients with acute MD onset had a better outcome than those with subacute-to-chronic MD onset (p = 0.008). Conclusions This study highlights the spectrum of CAS-associated with MD across the country. A progressive, age-dependent functional neuronal modulation in the basal ganglia due to CAS may underlie this condition.

Movement Disorders Associated With Cerebral Artery Stenosis: A Nationwide Study

A Case mimicking Neuroleptic Malignant Syndrome associated with Levodopa Withdrawal in Parkinson's Disease

We report a patient with a left anterior cerebral artery (ACA) infarct showing representational neglect. Representational neglect is some form of neglect to areas of imagined space. We believe that the description of our case gives further information about neglect syndrome and the functional asymmetry of the cerebral hemispheres because these observations are rare.

Representational Neglect in Left Anterior Cerebral Artery Infarction.

Acute neuropathic complications rarely develop in patients with inflammatory bowel disease. A case of acute peripheral neuropathy that developed only 4 weeks after a diagnosis of Crohn’s disease (CD) is reported herein. Peripheral neuropathy can be a rare extraintestinal manifestation of CD. Early recognition and proper management of the neurologic complications are crucial for preventing the associated morbidity. J Korean Neurol Assoc 31(4):258-261, 2013

Acute Peripheral Neuropathy, as One of Early Manifestations in Crohn’s Disease

Involuntary movement associated with deep watershed ischemic lesions has been rarely reported. A 67-year-old woman presented with acute hemichorea on the left side. Magnetic resonance imaging showed acute infarcts in the anterior border zone. On perfusion studies, impaired cerebral blood flow was observed on the subcortical region sparing the basal ganglia. Cerebral angiogram confirmed severe stenosis in the right internal carotid artery. Her hemichorea gradually improved along with normalization of perfusion after carotid artery stenting with angioplasty. We suggest that impaired cerebral blood flow in critical watershed territories may be an important contributing factor in hemichorea associated with carotid occlusive disease.

/pdf/acute-hemichorea-as-an-unusual-presentation-of-internal-2a9g94zmu5.pdf

Acute hemichorea as an unusual presentation of internal carotid artery stenosis.

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A Case mimicking Neuroleptic Malignant Syndrome associated with Levodopa Withdrawal in Parkinson's Disease

Representational Neglect in Left Anterior Cerebral Artery Infarction.

Acute Peripheral Neuropathy, as One of Early Manifestations in Crohn’s Disease

Acute hemichorea as an unusual presentation of internal carotid artery stenosis.