Peripheral Plasma Vitamin D and Non-HDL Cholesterol Reflect the Severity of Cerebral Cavernous Malformation Disease

Girard, Romuald; Khanna, Omaditya; Shenkar, Robert; Zhang, Lingjiao; Wu, Meijing; Jesselson, Michael; Zeineddine, Hussein A.; Gangal, Anupriya; Fam, Maged D; Gibson, Christopher C.; Whitehead, Kevin J.; Li, Dean Y.; Liao, James K.; Shi, Changbin

doi:10.2217/bmm.15.118

Cited by 38 publications

(41 citation statements)

References 38 publications

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“…Overall, it is plausible that the impaired endothelial function that may accompany low circulating vitamin D levels contributes to an increased risk of cerebrovascular diseases and mortality. Indeed, there is increasing interest in how vitamin D levels might influence the onset and severity of cerebrovascular diseases, including stroke [145][146][147][148][149][150] and cerebrovascular malformations [43,95,117,128,151,152].…”

Section: Vitamin D Deficiency and Its Impact On Cerebrovascular Diseasesmentioning

confidence: 99%

Vitamin D Deficiency and the Risk of Cerebrovascular Disease

Kim

Perrelli

Ragni³

et al. 2020

Antioxidants

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Vitamin D deficiency has been clearly linked to major chronic diseases associated with oxidative stress, inflammation, and aging, including cardiovascular and neurodegenerative diseases, diabetes, and cancer. In particular, the cardiovascular system appears to be highly sensitive to vitamin D deficiency, as this may result in endothelial dysfunction and vascular defects via multiple mechanisms. Accordingly, recent research developments have led to the proposal that pharmacological interventions targeting either vitamin D deficiency or its key downstream effects, including defective autophagy and abnormal pro-oxidant and pro-inflammatory responses, may be able to limit the onset and severity of major cerebrovascular diseases, such as stroke and cerebrovascular malformations. Here we review the available evidence supporting the role of vitamin D in preventing or limiting the development of these cerebrovascular diseases, which are leading causes of disability and death all over the world.

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Section: Vitamin D Deficiency and Its Impact On Cerebrovascular Diseasesmentioning

confidence: 99%

Vitamin D Deficiency and the Risk of Cerebrovascular Disease

Kim

Perrelli

Ragni³

et al. 2020

Antioxidants

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“…Correlation of clinical aggressiveness with biomarkers, determined either biologically or by using neuroimaging techniques, has also being progressively subjected to several studies [30,31,32] in order to better understand the distinct clinical behavior of the patients harboring CCMs. Main limitations of this study are the retrospective nature, small sample, and the fact that it was being originated from a reference center; these factors might not represent adequately the non-intervention CCM population.…”

Section: Resultsmentioning

confidence: 99%

Cerebral Cavernous Malformations: Patient-Reported Outcome Validates Conservative Management

et al. 2017

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Background: Cerebral cavernous malformations (CCM) are clusters of dilated sinusoidal channels lined by a single layer of endothelium. In contradistinction to arteriovenous malformations, these lesions do not have smooth muscle or elastin in their lining and they are angiographically occult, and the MRI is the most sensitive test for CCM detection. CCM are one of the most prevalent vascular malformations of the central nervous system, affecting about 0.4-0.6% of the general population. The main complication of this malformation is the risk of bleeding, which may cause neurological deficits that affect the quality of life (QoL) in patients. When symtomatic, they may be surgically treated for relieving the mass effect and seizures refractory to drug uses, hemorrhage and drug-refractory epilepsy. Patient-reported outcome (PRO) may be a strategy that can be used to evaluate QoL of CCM population and was used in a sample of non-operated patients. Methods: An observational, cross-sectional analysis to evaluate the PRO using the SF-36 and EuroQol 5 dimensions (EQ-5D) questionnaires of QoL added to functional metrics using the Karnofsky Performance Status (KPS) in 49 patients not submitted to intervention and with long-term follow-up. Results: During the 364 person-years of follow-up, there was an average of individual follow-up of 7.42 years. The mean age was 46.8 years (18-84) - 57% of them were female, 71% had superficial lesions, and 65% had the familial form. Comparisons of SF-36 dimensions with KPS graded <100 had a worse score only in terms of the pain (p = 0.04), vitality (p = 0.001), and general state of health (p = 0.03) domains. The domain mental health was worse in patients without surgical indication (p = 0.032). The functional capacity domain had the highest overall grading in the group. The EQ-5D dimensions of mobility (p = 0.03) and pain/discomfort (p = 0.001) were the ones with lower score compared to KPS <100. Conclusion: The study is the first to evaluate, with validated tools, the PRO of non-operated CCM patients and has demonstrated in a selected group of patients that it was possible to achieve long-term clinical stability, thereby maintaining QoL and functional neurological outcome.

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“…In an in vitro study by Gibson et al [2], vitamin D3 was found to restore structural abnormalities in human endothelial cells deficient in CCM2. In a clinical study by Girard et al [3], 43 CM patients underwent labora- tory testing to assess 25-hydroxy-vitamin D, approximately half of whom were familial form. Patients were stratified into "acute" and "chronic" aggressiveness.…”

Section: Discussionmentioning

confidence: 99%

“…Recently, vitamin D has been shown to play a role in CM endothelial leakiness in animal and in vitro models [2]. Only one study has assessed the influence of 25-hydroxy-vitamin D level on the clinical course of patients with CM [3]. In that study, lower 25-hydroxy-vitamin D was found in CM patients with "chronically aggressive disease" defined as one of the following: early age at symptom onset, 2 or more symptomatic hemorrhages, or high lesion burden.…”

Section: Introductionmentioning

confidence: 99%

Cavernous Malformation Hemorrhagic Presentation at Diagnosis Associated with Low 25-Hydroxy-Vitamin D Level

et al. 2020

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Background: Cavernous malformations (CM) are angiographically occult vascular malformations that may be incidental or present with intracerebral or spinal hemorrhage, seizures, or nonhemorrhagic focal neurologic deficit (FND). Recently in vitro data have suggested vitamin D may play a role in stabilizing CCM2 endothelial cells. Little is known about the effect of vitamin D in human CM disease. Methods: Beginning in 2015, consecutive patients at our institution with radiologically confirmed CM were recruited to participate in a prospective clinical registry as well as 25-hydroxy-vitamin D study. A structured interview, survey, and examination were performed at baseline. Medical records and magnetic resonance imaging studies were reviewed and data collected included comorbid conditions, medication use, and location of CM. Standard definition of clinical hemorrhage, FND, and seizures was used. Univariate and multivariate logistic regression models were used, and OR, 95% CIs, and likelihood-ratio p values were calculated to de-

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Peripheral Plasma Vitamin D and Non-HDL Cholesterol Reflect the Severity of Cerebral Cavernous Malformation Disease

Cited by 38 publications

References 38 publications

Vitamin D Deficiency and the Risk of Cerebrovascular Disease

Vitamin D Deficiency and the Risk of Cerebrovascular Disease

Cerebral Cavernous Malformations: Patient-Reported Outcome Validates Conservative Management

Cavernous Malformation Hemorrhagic Presentation at Diagnosis Associated with Low 25-Hydroxy-Vitamin D Level

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