Serial brain CT in corticobasal degeneration: radiological and pathological correlation of two autopsy cases

Tsuchiya, Kuniaki; Miyazaki, Hiroshi; Ikeda, Kenji; Watabiki, Sadakiyo; Kijima, Yoshimaro; Sano, Mizuka; Shimada, H

doi:10.1016/s0022-510x(97)00138-x

Cited by 12 publications

(3 citation statements)

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“…We found only 2 articles11, 12 that focused specifically on dystonia in CBD. Of the 404 selected cases, there were not sufficient data to conclude if there was dystonia in 108 (26.7%)14–30; of the remaining 296 cases, 111 (37.5%) were classified as CBD with dystonia,1, 2, 5, 10, 11, 31–67 and 185 (62.5%) were classified as CBD without dystonia1, 5, 10, 15, 18, 27, 28, 35, 36, 39, 40, 45, 47–50, 52, 55, 57, 57–59, 61, 62, 64, 66–80 (see Supplementary Tables 1 and 2, respectively, for all references). There was no significant difference between CBD with and without dystonia in age at onset (61.9 ± 16.5 vs 64.1 ± 6.7 years, respectively; P = .22), and disease duration (6.4 ± 2.7 vs 5.5 ± 2.6 years, respectively; P = .11).…”

Section: Resultsmentioning

confidence: 99%

Dystonia in corticobasal degeneration: A review of the literature on 404 pathologically proven cases

2012

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A B S T R A C T :Dystonia is considered one of the classical features of corticobasal degeneration and is reported in up to 83% in clinical, not pathologically confirmed, series. Here, we aimed to establish the frequency and the clinical characteristics of dystonia in CBD by reviewing the literature on 404 pathologically proven cases. Further, we aimed to identify the frequency and characteristics of dystonia in all described phenotypes with CBD pathology. Dystonia was present in only 37.5% of the 296 cases with adequate information. The majority of the cases with dystonia presented with a corticobasal syndrome, and dystonia occurred in the first 2 years from disease onset, affecting the upper limb. In cases with dystonia that presented with a ''dementia'' phenotype, dystonia tended to appear later in the disease course and to more affect the cervical region and the face. With regard to the distribution of the phenotypes, fifty-four percent of 374 cases presented as corticobasal syndrome, 15% as frontotemporal dementia, and 10.7% as progressive supranuclear palsy. Dystonia and myoclonus were present in about half of all cases with corticobasal syndrome, implying that these features may not be as frequent in corticobasal syndrome as are akinetic-rigid syndrome and apraxia (100% and 86.3%, respectively). Dystonia and myoclonus almost co-occurred in our analysis, suggesting a possible association. In conclusion, despite dystonia being an inclusion criterion in all sets of clinical criteria for corticobasal degeneration, this was present in only one third of the pathologically proven cases presented here. More accurate characterization of dystonia in corticobasal degeneration would be of importance for clinical diagnosis and development of treatment strategies. V C 2012 Movement Disorder Society

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Section: Resultsmentioning

confidence: 99%

Dystonia in corticobasal degeneration: A review of the literature on 404 pathologically proven cases

2012

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“…Glucose hypometabolism of atrophy, although one study did find atrophy in the head of the caudate nuclei. 49 However, studies with FDG PET found that CBD presents reduced glucose metabolism in the frontal lobe and in the subcortical structures such as thalamus, caudate and putamen with marked asymmetry between the two hemispheres. Based on these observations, we may hypothesize that the neurobiological process of these diseases might interact with a brain morphology conferring a specific vulnerability for diseases of the FTLD spectrum in the H1 carriers.…”

Section: Discussionmentioning

confidence: 99%

H1 haplotype of the MAPT gene is associated with lower regional gray matter volume in healthy carriers

et al. 2008

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The microtubule-associated protein Tau (MAPT) gene codes for the protein Tau that is involved in the pathogenesis of neurodegenerative diseases. Recent studies have detected an over-representation of the H1 haplotype of the MAPT gene in neurodegenerative disorders such as progressive supranuclear palsy (PSP), corticobasal degeneration (CBD), frontotemporal dementia (FTD) and Parkinson's disease (PD), whereas the H2 haplotype has been found to be related to familial FTD. We aimed to investigate the association between MAPT haplotype status and brain morphology in healthy adults. A total of 150 healthy subjects underwent 3D high-resolution magnetic resonance (MR). MR images were processed following an optimized protocol to perform the Voxel-based morphometry (VBM) comparisons of the gray matter (GM) in H1 carriers (n ¼ 141) in contrast to H2H2 homozygous (n ¼ 9), and H1H1 homozygous (n ¼ 85) in contrast to H2 carriers (n ¼ 65). The threshold for statistical significance was 0.005 uncorrected. Opposite comparisons were also carried out. The groups had similar demographic and cognitive features. Compared with H2H2, the H1 carriers showed up to 19% smaller GM volume in the head of the right caudate nucleus, in the right middle frontal gyrus, in the left insula and orbito-frontal cortex, and in the inferior temporal and inferior cerebellar lobes, bilaterally. Compared with all H2 carriers, H1H1 displayed lower GM in the same regions, but the effect was smaller (5%), possibly due to a dilution effect by H1 in the H2 carriers group. The data suggest that H1 haplotype is associated with a particular cerebral morphology that may increase the susceptibility of the healthy carriers to develop neurodegenerative diseases such as sporadic tauopathies.

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“…According to Kertesz et al. ‘Pick complex’ is composed of various degenerative diseases and clinical syndromes, including Pick's disease with Pick bodies (PDPB), corticobasal degeneration (CBD), generalized variant of Pick's disease (gvPD), frontotemporal dementia, dementia lacking distinctive histology (DLDH), frontotemporal dementia and parkinsonism linked to chromore 17 (FTDP‐17), progressive subcortical gliosis (PSG), amyotrophic lateral sclerosis with dementia (ALS with dementia), primary progressive aphasia (PPA), semantic dementia, and primary progressive apraxia 5–15 …”

Section: Introductionmentioning

confidence: 99%

Basal ganglia lesions in ‘Pick complex’: A topographic neuropathological study of 19 autopsy cases

Tsuchiya

Ikeda

2002

Neuropathology

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The distribution of basal ganglia lesions, including the amygdala, striatum (caudate nucleus, putamen), pallidum, and substantia nigra, were reinvestigated in 19 Japanese autopsy cases of 'Pick complex', consisting of five patients with corticobasal degeneration (CBD), 10 patients with Pick's disease with Pick bodies (PDPB), and four patients with generalized variant of Pick's disease (gvPD). The lesions in the amygdala, striatum, and pallidum were classified into three categories (severe, moderate, and slight). The lesions in the substantia nigra were qualitatively judged, compared with normal controls. In CBD, basal ganglia lesions in all five cases were uniform: the pallidum showed severe lesions, the striatum moderate lesions, the amygdala slight lesions, and obvious neuronal loss of the substantia nigra was verified in all five cases. Basal ganglia lesions in 10 cases of PDPB were also uniform: the amygdala disclosed severe to moderate lesions, the striatum moderate to slight lesions, the pallidum slight lesions, while obvious neuronal loss of the substantia nigra was found in only two of nine cases in which this structure was examined. Furthermore, basal ganglia lesions in all four cases of gvPD were uniform: the caudate nucleus showed severe lesions, the putamen and amygdala severe to moderate lesions, the pallidum moderate to slight lesions, and obvious neuronal loss of the substantia nigra was confirmed in all four cases. This study, using conventional staining such as hematoxylin-eosin and Holzer, clarified that there were prominent lesions in the pallidum in CBD, in the amygdala in PDPB, and in the caudate nucleus in gvPD, respectively. In addition, nigral involvement was usually found in CBD and gvPD, but was rarely seen in PDPB. These neuropathological findings may help to elucidate the pathological heterogeneity of basal ganglia lesions in 'Pick complex'.

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Serial brain CT in corticobasal degeneration: radiological and pathological correlation of two autopsy cases

Cited by 12 publications

References 10 publications

Dystonia in corticobasal degeneration: A review of the literature on 404 pathologically proven cases

Dystonia in corticobasal degeneration: A review of the literature on 404 pathologically proven cases

H1 haplotype of the MAPT gene is associated with lower regional gray matter volume in healthy carriers

Basal ganglia lesions in ‘Pick complex’: A topographic neuropathological study of 19 autopsy cases

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