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Structural and functional MRI abnormalities of cerebellar cortex and nuclei in SCA3, SCA6 and Friedreich's ataxia.
Stefanescu, Maria R; Dohnalek, Moritz; Maderwald, Stefan; Thürling, Markus; Minnerop, Martina; Beck, Andreas; Schlamann, Marc; Diedrichsen, Joern; Ladd, Mark E; Timmann, Dagmar.
  • Stefanescu MR; 1 Department of Neurology, University of Duisburg-Essen, Essen, Germany 2 Erwin L. Hahn Institute for Magnetic Resonance Imaging, University of Duisburg-Essen, Essen, Germany stefanescu.m.roxana@gmail.com.
  • Dohnalek M; 1 Department of Neurology, University of Duisburg-Essen, Essen, Germany.
  • Maderwald S; 2 Erwin L. Hahn Institute for Magnetic Resonance Imaging, University of Duisburg-Essen, Essen, Germany.
  • Thürling M; 1 Department of Neurology, University of Duisburg-Essen, Essen, Germany 2 Erwin L. Hahn Institute for Magnetic Resonance Imaging, University of Duisburg-Essen, Essen, Germany.
  • Minnerop M; 3 Institute of Neuroscience and Medicine (INM-1), Research Centre Jülich, Jülich, Germany 4 Department of Neurology, University of Bonn, Bonn, Germany.
  • Beck A; 5 Department of Computer Sciences, University of Düsseldorf, Düsseldorf, Germany.
  • Schlamann M; 6 Department of Diagnostic and Interventional Radiology and Neuroradiology, University of Duisburg-Essen, Essen, Germany.
  • Diedrichsen J; 7 Institute of Cognitive Neuroscience, University College London, London, UK.
  • Ladd ME; 2 Erwin L. Hahn Institute for Magnetic Resonance Imaging, University of Duisburg-Essen, Essen, Germany 6 Department of Diagnostic and Interventional Radiology and Neuroradiology, University of Duisburg-Essen, Essen, Germany 8 Division of Medical Physics in Radiology, University of Heidelberg and Ger
  • Timmann D; 1 Department of Neurology, University of Duisburg-Essen, Essen, Germany.
Brain ; 138(Pt 5): 1182-97, 2015 May.
Article en En | MEDLINE | ID: mdl-25818870
Spinocerebellar ataxia type 3, spinocerebellar ataxia type 6 and Friedreich's ataxia are common hereditary ataxias. Different patterns of atrophy of the cerebellar cortex are well known. Data on cerebellar nuclei are sparse. Whereas cerebellar nuclei have long been thought to be preserved in spinocerebellar ataxia type 6, histology shows marked atrophy of the nuclei in Friedreich's ataxia and spinocerebellar ataxia type 3. In the present study susceptibility weighted imaging was used to assess atrophy of the cerebellar nuclei in patients with spinocerebellar ataxia type 6 (n = 12, age range 41-76 years, five female), Friedreich's ataxia (n = 12, age range 21-55 years, seven female), spinocerebellar ataxia type 3 (n = 10, age range 34-67 years, three female), and age- and gender-matched controls (total n = 23, age range 22-75 years, 10 female). T1-weighted magnetic resonance images were used to calculate the volume of the cerebellum. In addition, ultra-high field functional magnetic resonance imaging was performed with optimized normalization methods to assess function of the cerebellar cortex and nuclei during simple hand movements. As expected, the volume of the cerebellum was markedly reduced in spinocerebellar ataxia type 6, preserved in Friedreich's ataxia, and mildy reduced in spinocerebellar ataxia type 3. The volume of the cerebellar nuclei was reduced in the three patient groups compared to matched controls (P-values < 0.05; two-sample t-tests). Atrophy of the cerebellar nuclei was most pronounced in spinocerebellar ataxia type 6. On a functional level, hand-movement-related cerebellar activation was altered in all three disorders. Within the cerebellar cortex, functional magnetic resonance imaging signal was significantly reduced in spinocerebellar ataxia type 6 and Friedreich's ataxia compared to matched controls (P-values < 0.001, bootstrap-corrected cluster-size threshold; two-sample t-tests). The difference missed significance in spinocerebellar ataxia type 3. Within the cerebellar nuclei, reductions were significant when comparing spinocerebellar ataxia type 6 and Friedreich's ataxia to matched controls (P < 0.01, bootstrap-corrected cluster-size threshold; two-sample t-tests). Susceptibility weighted imaging allowed depiction of atrophy of the cerebellar nuclei in patients with Friedreich's ataxia and spinocerebellar ataxia type 3. In spinocerebellar ataxia type 6, pathology was not restricted to the cerebellar cortex but also involved the cerebellar nuclei. Functional magnetic resonance imaging data, on the other hand, revealed that pathology in Friedreich's ataxia and spinocerebellar ataxia type 3 is not restricted to the cerebellar nuclei. There was functional involvement of the cerebellar cortex despite no or little structural changes.
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Texto completo: 1 Banco de datos: MEDLINE Asunto principal: Ataxia de Friedreich / Imagen por Resonancia Magnética / Corteza Cerebelosa / Núcleos Cerebelosos / Enfermedad de Machado-Joseph / Ataxias Espinocerebelosas Límite: Adult / Aged / Female / Humans / Male / Middle aged Idioma: En Año: 2015 Tipo del documento: Article

Texto completo: 1 Banco de datos: MEDLINE Asunto principal: Ataxia de Friedreich / Imagen por Resonancia Magnética / Corteza Cerebelosa / Núcleos Cerebelosos / Enfermedad de Machado-Joseph / Ataxias Espinocerebelosas Límite: Adult / Aged / Female / Humans / Male / Middle aged Idioma: En Año: 2015 Tipo del documento: Article