Blood–brain barrier breakdown in reversible cerebral vasoconstriction syndrome: Implications for pathophysiology and diagnosis

Lee, Mi Ji; Cha, Jihoon; Choi, Hae Woon; Woo, Sook Young; Kim, Seonwoo; Wang, Shuu Jiun; Chung, Chin Sang

doi:10.1002/ana.24891

Cited by 96 publications

(83 citation statements)

References 54 publications

(194 reference statements)

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“…1,12 Some of these mechanisms have also been hypothesized in RCVS. [13][14][15] In conclusion we can assume that SLE, notably SLE flare-up as observed in our patients, or SLE drugs, may represent a particular risk factor for RCVS. The observation of a positive familial history for SLEfour affected relativesin Patient 1 and an abnormal short stature in Patient 2 might also suggest underlying predisposing factors for both SLE and RCVS.…”

Section: Discussionsupporting

confidence: 50%

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Reversible cerebral vasoconstriction syndrome in paediatric patients with systemic lupus erythematosus: implications for management

Durrleman

Naggara

Grévent

et al. 2018

Develop Med Child Neuro

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Microvascular brain injury is well recognized in neuropsychiatric systemic lupus erythematosus (SLE), but cerebral large artery involvement is being debated. Three females with SLE, aged 9 to 14 years, had immunosuppressive treatment intensification because of lupus nephritis. Within the following days or weeks, they presented with intense cephalalgia – isolated or associated with neurological symptoms – and no or mild hypertension. Magnetic resonance angiography showed multiple stenoses within the circle of Willis. One patient had subsequent small subcortical cerebral infarction. Two patients were treated for neuropsychiatric SLE; one patient was treated for reversible cerebral vasoconstriction syndrome (RCVS). Angiography normalized within a few weeks in all three patients. Retrospectively, clinical and radiological features suggest that RCVS was the most likely diagnosis in all patients. Multidisciplinary analysis of clinical and angiographic features is recommended, as RCVS is rare in children and its recognition may help to adjust treatment. What this paper adds Reversible vasoconstriction syndrome was observed in paediatric systemic lupus erythematosus. Thorough imaging analysis was necessary to address this diagnosis in paediatric patients.

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Section: Discussionsupporting

confidence: 50%

“…Another mechanism involves autoimmune inflammation injury, mediated by autoantibody production, complement activation, increased permeability of the blood–brain barrier, intrathecal production of proinflammatory cytokines, and immune complexes . Some of these mechanisms have also been hypothesized in RCVS …”

Section: Discussionmentioning

confidence: 99%

Reversible cerebral vasoconstriction syndrome in paediatric patients with systemic lupus erythematosus: implications for management

Durrleman

Naggara

Grévent

et al. 2018

Develop Med Child Neuro

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“…BBB damage is a common pathological feature shared by stroke and a variety of neurological diseases . Notably, it is closely associated with poor neurological outcomes .…”

Section: Introductionmentioning

confidence: 99%

The peripheral immune response after stroke—A double edge sword for blood‐brain barrier integrity

Zhu

Huang

et al. 2018

CNS Neurosci Ther

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Summary The blood‐brain barrier (BBB) is a highly regulated interface that separates the peripheral circulation and the brain. It plays a vital role in regulating the trafficking of solutes, fluid, and cells at the blood‐brain interface and maintaining the homeostasis of brain microenvironment for normal neuronal activity. Growing evidence has led to the realization that ischemic stroke elicits profound immune responses in the circulation and the activation of multiple subsets of immune cells, which in turn affect both the early disruption and the later repair of the BBB after stroke. Distinct phenotypes or subsets of peripheral immune cells along with diverse intracellular mechanisms contribute to the dynamic changes of BBB integrity after stroke. This review focuses on the interaction between the peripheral immune cells and the BBB after ischemic stroke. Understanding their reciprocal interaction may generate new directions for stroke research and may also drive the innovation of easy accessible immune modulatory treatment strategies targeting BBB in the pursuit of better stroke recovery.

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“…[5][6][7][8][9][10][11][12][13] Patients with RCVS are at risk of severe complications including posterior reversible encephalopathy syndrome, ischemic stroke, and intracranial hemorrhage (including cortical subarachnoid, intracerebral, and subdural hemorrhage). [5][6][7][8][9][10]14 Sympathetic overactivity, 15 oxidative stress, 16 an impaired endothelial repair capacity, 17 and genetic predisposition 18 may contribute to the dysregulated vascular tone of cerebral arteries in RCVS, 4,19,20 though a definitive pathogenesis has not yet been delineated.…”

Section: Introductionmentioning

confidence: 99%

Influence of Climate on the Incidence of RCVS – A Retrospective Study From Taiwan

et al. 2019

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Background Cold weather is reportedly a precipitator of reversible cerebral vasoconstriction syndrome (RCVS) in a few cases. We systematically investigated whether meteorological factors correlate with the occurrence of RCVS. Methods We conducted a retrospective analysis of a cohort of patients diagnosed with RCVS or probable RCVS, based on International Classification of Headache Disorders, third edition (ICHD‐3) criteria, in a hospital‐based headache center from March 2005 to February 2014. Monthly averages of local weather data measured in Taipei were obtained from the Central Weather Bureau in Taiwan. Primary weather variables were compared with the number of monthly new‐onset cases of RCVS. Results We recruited 226 patients with established RCVS and 72 patients with probable RCVS during a 108‐month study period. Incidence of RCVS was higher in winter than summer months (3.3 persons/month [SD: 2.0] vs 2.1 persons/month [SD: 1.5], P = .013). The monthly incidence of RCVS correlated negatively with mean daily temperature (r = −0.231, P = .016) and average precipitation (r = −0.269, P = .005), but positively with barometric pressure (r = 0.274, P = .004). These 3 correlated meteorological factors together explained about 10% of the variance in RCVS monthly incidence (R2 = 0.095, P = .015). Conclusion RCVS was found to be more common in winter months and to be associated with weather variables in Taiwan. Further studies are needed to explore the underlying mechanisms of these associations.

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Blood–brain barrier breakdown in reversible cerebral vasoconstriction syndrome: Implications for pathophysiology and diagnosis

Abstract: This is the first study to show BBB breakdown in patients with RCVS. This finding might broaden our understanding of the pathophysiology and clinical spectrum of RCVS. Ann Neurol 2017;81:454-466.

Cited by 96 publications

References 54 publications

Reversible cerebral vasoconstriction syndrome in paediatric patients with systemic lupus erythematosus: implications for management

Reversible cerebral vasoconstriction syndrome in paediatric patients with systemic lupus erythematosus: implications for management

The peripheral immune response after stroke—A double edge sword for blood‐brain barrier integrity

Influence of Climate on the Incidence of RCVS – A Retrospective Study From Taiwan

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