Diffusion tensor imaging of the extracorticospinal network in the brains of patients with Wilson disease

Zhou, Xiangxue; Li, Xunhua; Qin, Haolin; Li, Gui-dian; Huang, Hongbo; Liang, Yingying; Liang, Xiaoyan; Pu, Xiao‐Yong

doi:10.1016/j.jns.2016.02.006

Cited by 12 publications

(13 citation statements)

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“…WD is an autosomal recessive defect of cellular copper export. Neurological symptoms of WD is secondary to damage to networks of the extracorticospinal tract, which includes both nuclei and fiber projections (Jadav et al., 2013; King, Walshe, Kendall, Halligan, & Hall‐Craggs, 1996; Prashanth et al., 2010; Xiang‐xue et al., 2016a). The motor asymmetry in WD may be related to asymmetry of copper deposition and damage to extracorticospinal tract.…”

Section: Discussionmentioning

confidence: 99%

The asymmetry of neural symptoms in Wilson's disease patients detecting by diffusion tensor imaging, resting‐state functional MRI, and susceptibility‐weighted imaging

Zhou

Chen

et al. 2018

Brain and Behavior

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ObjectiveTo investigate the cause of the motor asymmetry in Wilson's disease (WD) patients using functional MRI.MethodsFifty patients with WD and 20 age‐matched healthy controls were enrolled. Neurological symptoms were scored using the modified Young Scale. All study subjects underwent diffusion tensor imaging (DTI), susceptibility‐weighted imaging (SWI), and resting‐state functional MRI (rs‐fMRI) of the brain. Six regions of interest (ROI) were chosen. Fiber volumes between ROIs on DTI, corrected phase (CP) values on SWI, amplitude of low‐frequency fluctuation (ALFF), and regional homogeneity (REHO) values on rs‐fMRI were determined. Asymmetry index (right or left value/left or right value) was evaluated.ResultsAsymmetry of rigidity, tremor, choreic movement, and gait abnormality (asymmetry index = 1.33, 1.39, 1.36, 1.40), fiber tracts between the GP and substantia nigra (SN), GP and PU, SN and thalamus (TH), SN and cerebellum, head of the caudate nucleus (CA) and SN, PU and CA, CA and TH, TH and cerebellum (asymmetry index = 1.233, 1.260, 1.269, 1.437, 1.503, 1.138, 1.145, 1.279), CP values in the TH, SN (asymmetry index = 1.327, 1.166), ALFF values, and REHO values of the TH (asymmetry index = 1.192, 1.233) were found. Positive correlation between asymmetry index of rigidity and fiber volumes between the GP and SN, SN and TH (r = .221, .133, p = .043, .036), and tremor and fiber volumes between the CA and TH (r = .045, p = .040) was found.ConclusionsThe neurological symptoms of patients with WD were asymmetry. The asymmetry of fiber projections may be the main cause of motor asymmetry in patients with WD.

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Section: Discussionmentioning

confidence: 99%

The asymmetry of neural symptoms in Wilson's disease patients detecting by diffusion tensor imaging, resting‐state functional MRI, and susceptibility‐weighted imaging

Zhou

Chen

et al. 2018

Brain and Behavior

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“…Increased 64 Cu radioactivity was detected in all of brain regions in Atp7b −/− knockout mice by 21 th week of age, including regions of cortex, basal ganglia, thalamus, midbrain and brain stem and cerebellum, with highest 64 Cu radioactivity detected in cerebellum [Fig 5]. It will be useful to correlate 64 Cu radioactivity detected by [ 64 Cu]CuCl 2 PET/CT with abnormalities detected by other imaging modalities, such as abnormal fiber projection between subcortical nuclei [31], for delineation of molecular mechanism of brain tissue damage in WD patients with neuropsychiatric disorders.…”

Section: Discussionmentioning

confidence: 99%

Age-dependent changes of cerebral copper metabolism in Atp7b −/− knockout mouse model of Wilson’s disease by [64Cu]CuCl2-PET/CT

Xie

Pascual

et al. 2017

Metab Brain Dis

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Copper is a nutritional metal required for brain development and function. Wilson’s disease (WD), or hepatolenticular degeneration, is an inherited human copper metabolism disorder caused by mutation of ATP7B gene. Many WD patients present with variable neurological and psychiatric symptoms, which may be related to neurodegeneration secondary to copper metabolism imbalance. The objective of this study is to explore feasibility and use of copper-64 chloride ([64C]CuCl2) as a tracer for noninvasive assessment of age-dependence changes of cerebral copper metabolism in WD using an Atp7b−/− knockout mouse model of WD and a positron emission tomography/computed tomography (PET/CT) scanner. Continuing from recent study of biodistribution and radiation dosimetry of [64C]CuCl2 in Atp7b−/− knockout mice, PET quantitative analysis revealed low 64Cu radioactivity in the brains of Atp7b−/− knockout mice at 7th week of age, compared with the 64Cu radioactivity in the brains of age and gender-matched wild type C57BL/6 mice, at 24 hour (h) post intravenous injection of [64C]CuCl2 as a tracer. Furthermore, age-dependent increase of 64Cu radioactivity was detected in the brains of Atp7b−/− knockout mice from 13th to 21th week of age, using the data derived from a longitudinal [64C]CuCl2-PET/CT study of Atp7b−/− knockout mice with orally administered [64Cu]CuCl2 as a tracer. The findings of this study support the use of [64Cu]CuCl2-PET/CT as a tool for noninvasive assessment of age-dependent changes of cerebral copper metabolism in WD patients presenting with variable neurological and psychiatric symptoms.

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“…Diffusion tensor imaging (DTI) is the most common dMRI technique. Studies have reported decreased fractional anisotropy (FA) and increased mean diffusivity (MD) in white matter and BG in Wilson's disease …”

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A study of neurite orientation dispersion and density imaging in wilson's disease

Song

Huang

et al. 2017

Magnetic Resonance Imaging

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Diffusion tensor imaging of the extracorticospinal network in the brains of patients with Wilson disease

Abstract: DTI can reconstruct the network of the extracorticospinal tract. Fiber projection between subcortical nuclei was abnormal in WD patients. Damage to fiber connections may correlate with neurological symptoms in WD patients.

Cited by 12 publications

References 22 publications

The asymmetry of neural symptoms in Wilson's disease patients detecting by diffusion tensor imaging, resting‐state functional MRI, and susceptibility‐weighted imaging

The asymmetry of neural symptoms in Wilson's disease patients detecting by diffusion tensor imaging, resting‐state functional MRI, and susceptibility‐weighted imaging

Age-dependent changes of cerebral copper metabolism in Atp7b −/− knockout mouse model of Wilson’s disease by [64Cu]CuCl2-PET/CT

A study of neurite orientation dispersion and density imaging in wilson's disease

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