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Quantitative MRI of the spinal cord and brain in adrenomyeloneuropathy: in vivo assessment of structural changes.

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  • Additional Information
    • Publication Year:
      2016
    • NAICS/Industry Codes:
      NAICS/Industry Codes 621512 Diagnostic Imaging Centers
    • Abstract:
      Adrenomyeloneuropathy is the late-onset form of X-linked adrenoleukodystrophy, and is considered the most frequent metabolic hereditary spastic paraplegia. In adrenomyeloneuropathy the spinal cord is the main site of pathology. Differently from quantitative magnetic resonance imaging of the brain, little is known about the feasibility and utility of advanced neuroimaging in quantifying the spinal cord abnormalities in hereditary diseases. Moreover, little is known about the subtle pathological changes that can characterize the brain of adrenomyeloneuropathy subjects in the early stages of the disease. We performed a cross-sectional study on 13 patients with adrenomyeloneuropathy and 12 age-matched healthy control subjects who underwent quantitative magnetic resonance imaging to assess the structural changes of the upper spinal cord and brain. Total cord areas from C2-3 to T2-3 level were measured, and diffusion tensor imaging metrics, i.e. fractional anisotropy, mean, axial and radial diffusivity values were calculated in both grey and white matter of spinal cord. In the brain, grey matter regions were parcellated with Freesurfer and average volume and thickness, and mean diffusivity and fractional anisotropy from co-registered diffusion maps were calculated in each region. Brain white matter diffusion tensor imaging metrics were assessed using whole-brain tract-based spatial statistics, and tractography-based analysis on corticospinal tracts. Correlations among clinical, structural and diffusion tensor imaging measures were calculated. In patients total cord area was reduced by 26.3% to 40.2% at all tested levels (P < 0.0001). A mean 16% reduction of spinal cord white matter fractional anisotropy (P ≤ 0.0003) with a concomitant 9.7% axial diffusivity reduction (P < 0.009) and 34.5% radial diffusivity increase (P < 0.009) was observed, suggesting co-presence of axonal degeneration and demyelination. Brain tract-based spatial statistics showed a marked reduction of fractional anisotropy, increase of radial diffusivity (P < 0.001) and no axial diffusivity changes in several white matter tracts, including corticospinal tracts and optic radiations, indicating predominant demyelination. Tractography-based analysis confirmed the results within corticospinal tracts. No significant cortical volume and thickness reduction or grey matter diffusion tensor imaging values alterations were observed in patients. A correlation between radial diffusivity and disease duration along the corticospinal tracts (r = 0.806, P < 0.01) was found. In conclusion, in adrenomyeloneuropathy patients quantitative magnetic resonance imaging-derived measures identify and quantify structural changes in the upper spinal cord and brain which agree with the expected histopathology, and suggest that the disease could be primarily caused by a demyelination rather than a primitive axonal damage. The results of this study may also encourage the employment of quantitative magnetic resonance imaging in other hereditary diseases with spinal cord involvement. [ABSTRACT FROM AUTHOR]
    • Abstract:
      Copyright of Brain: A Journal of Neurology is the property of Oxford University Press / USA and its content may not be copied or emailed to multiple sites or posted to a listserv without the copyright holder's express written permission. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract. (Copyright applies to all Abstracts.)
    • ISSN:
      00068950
    • Accession Number:
      122969870
  • Citations
    • ABNT:
      CASTELLANO, A. et al. Quantitative MRI of the spinal cord and brain in adrenomyeloneuropathy: in vivo assessment of structural changes. Brain: A Journal of Neurology, [s. l.], v. 139, n. 6, p. 1735, 2016. Disponível em: http://search.ebscohost.com/login.aspx?direct=true&site=eds-live&db=edb&AN=122969870. Acesso em: 26 out. 2020.
    • AMA:
      Castellano A, Papinutto N, Cadioli M, et al. Quantitative MRI of the spinal cord and brain in adrenomyeloneuropathy: in vivo assessment of structural changes. Brain: A Journal of Neurology. 2016;139(6):1735. Accessed October 26, 2020. http://search.ebscohost.com/login.aspx?direct=true&site=eds-live&db=edb&AN=122969870
    • APA:
      Castellano, A., Papinutto, N., Cadioli, M., Brugnara, G., Iadanza, A., Scigliuolo, G., Pareyson, D., Uziel, G., Köhler, W., Aubourg, P., Falini, A., Henry, R. G., Politi, L. S., & Salsano, E. (2016). Quantitative MRI of the spinal cord and brain in adrenomyeloneuropathy: in vivo assessment of structural changes. Brain: A Journal of Neurology, 139(6), 1735.
    • Chicago/Turabian: Author-Date:
      Castellano, Antonella, Nico Papinutto, Marcello Cadioli, Gianluca Brugnara, Antonella Iadanza, Graziana Scigliuolo, Davide Pareyson, et al. 2016. “Quantitative MRI of the Spinal Cord and Brain in Adrenomyeloneuropathy: In Vivo Assessment of Structural Changes.” Brain: A Journal of Neurology 139 (6): 1735. http://search.ebscohost.com/login.aspx?direct=true&site=eds-live&db=edb&AN=122969870.
    • Harvard:
      Castellano, A. et al. (2016) ‘Quantitative MRI of the spinal cord and brain in adrenomyeloneuropathy: in vivo assessment of structural changes’, Brain: A Journal of Neurology, 139(6), p. 1735. Available at: http://search.ebscohost.com/login.aspx?direct=true&site=eds-live&db=edb&AN=122969870 (Accessed: 26 October 2020).
    • Harvard: Australian:
      Castellano, A, Papinutto, N, Cadioli, M, Brugnara, G, Iadanza, A, Scigliuolo, G, Pareyson, D, Uziel, G, Köhler, W, Aubourg, P, Falini, A, Henry, RG, Politi, LS & Salsano, E 2016, ‘Quantitative MRI of the spinal cord and brain in adrenomyeloneuropathy: in vivo assessment of structural changes’, Brain: A Journal of Neurology, vol. 139, no. 6, p. 1735, viewed 26 October 2020, .
    • MLA:
      Castellano, Antonella, et al. “Quantitative MRI of the Spinal Cord and Brain in Adrenomyeloneuropathy: In Vivo Assessment of Structural Changes.” Brain: A Journal of Neurology, vol. 139, no. 6, June 2016, p. 1735. EBSCOhost, search.ebscohost.com/login.aspx?direct=true&site=eds-live&db=edb&AN=122969870.
    • Chicago/Turabian: Humanities:
      Castellano, Antonella, Nico Papinutto, Marcello Cadioli, Gianluca Brugnara, Antonella Iadanza, Graziana Scigliuolo, Davide Pareyson, et al. “Quantitative MRI of the Spinal Cord and Brain in Adrenomyeloneuropathy: In Vivo Assessment of Structural Changes.” Brain: A Journal of Neurology 139, no. 6 (June 2016): 1735. http://search.ebscohost.com/login.aspx?direct=true&site=eds-live&db=edb&AN=122969870.
    • Vancouver/ICMJE:
      Castellano A, Papinutto N, Cadioli M, Brugnara G, Iadanza A, Scigliuolo G, et al. Quantitative MRI of the spinal cord and brain in adrenomyeloneuropathy: in vivo assessment of structural changes. Brain: A Journal of Neurology [Internet]. 2016 Jun [cited 2020 Oct 26];139(6):1735. Available from: http://search.ebscohost.com/login.aspx?direct=true&site=eds-live&db=edb&AN=122969870