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Cerebrovascular Phenotype in Fabry Disease Patients Assessed by Ultrasound

The result's identifiers

  • Result code in IS VaVaI

    <a href="https://www.isvavai.cz/riv?ss=detail&h=RIV%2F00064165%3A_____%2F23%3A10466010" target="_blank" >RIV/00064165:_____/23:10466010 - isvavai.cz</a>

  • Alternative codes found

    RIV/00216208:11110/23:10466010

  • Result on the web

    <a href="https://verso.is.cuni.cz/pub/verso.fpl?fname=obd_publikace_handle&handle=pIs1v7CxqN" target="_blank" >https://verso.is.cuni.cz/pub/verso.fpl?fname=obd_publikace_handle&handle=pIs1v7CxqN</a>

  • DOI - Digital Object Identifier

    <a href="http://dx.doi.org/10.1002/jum.16254" target="_blank" >10.1002/jum.16254</a>

Alternative languages

  • Result language

    angličtina

  • Original language name

    Cerebrovascular Phenotype in Fabry Disease Patients Assessed by Ultrasound

  • Original language description

    Objectives: Fabry disease (FD) is a rare X-linked lysosomal storage disorder with variable phenotypes, including neurological symptoms. These can be influenced by vascular impairment. Extracranial and transcranial vascular sonography is an effective and noninvasive method for measuring arterial structures and blood flow. The study aims to investigate cerebrovascular phenotype characteristics in FD patients compared to controls using neurosonology. Methods: This is a single-center, cross-sectional study of 130 subjects-65 patients (38 females), with genetically confirmed FD, and 65 sex- and age-matched controls. Using ultrasonography, we measured structural and hemodynamic parameters, including distal common carotid artery intima-media thickness, inner vertebral artery diameter, resting blood flow velocity, pulsatility index, and cerebral vasoreactivity (CVR) in the middle cerebral artery. To assess differences between FD and controls and to identify factors influencing investigated outcomes, unadjusted and adjusted regression analyses were performed. Results: In comparison to sex- and age-matched controls, FD patients displayed significantly increased carotid artery intima-media thickness (observed FD 0.69 +- 0.13 mm versus controls 0.63 +- 0.12 mm; Padj = .0014), vertebral artery diameter (observed FD 3.59 +- 0.35 mm versus controls 3.38 +- 0.33 mm; Padj = .0002), middle cerebral artery pulsatility index (observed FD 0.98 +- 0.19 versus controls 0.87 +- 0.11; Padj &lt; .0001), and significantly decreased CVR (observed FD 1.21 +- 0.49 versus controls 1.35 +- 0.38; Padj = .0409), when adjusted by age, BMI, and sex. Additionally, FD patients had significantly more variable CVR (0.48 +- 0.25 versus 0.21 +- 0.14; Padj &lt; .0001). Conclusions: Our results suggest the presence of multiple vascular abnormalities and changes in hemodynamic parameters of cerebral arteries in patients with FD.

  • Czech name

  • Czech description

Classification

  • Type

    J<sub>imp</sub> - Article in a specialist periodical, which is included in the Web of Science database

  • CEP classification

  • OECD FORD branch

    30103 - Neurosciences (including psychophysiology)

Result continuities

  • Project

    <a href="/en/project/LX22NPO5107" target="_blank" >LX22NPO5107: National institute for Neurological Research</a><br>

  • Continuities

    I - Institucionalni podpora na dlouhodoby koncepcni rozvoj vyzkumne organizace

Others

  • Publication year

    2023

  • Confidentiality

    S - Úplné a pravdivé údaje o projektu nepodléhají ochraně podle zvláštních právních předpisů

Data specific for result type

  • Name of the periodical

    Journal of Ultrasound in Medicine

  • ISSN

    0278-4297

  • e-ISSN

    1550-9613

  • Volume of the periodical

    42

  • Issue of the periodical within the volume

    10

  • Country of publishing house

    US - UNITED STATES

  • Number of pages

    16

  • Pages from-to

    2315-2330

  • UT code for WoS article

    000993186100001

  • EID of the result in the Scopus database

    2-s2.0-85159645731