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Longitudinal in vivo MRI in a Huntington’s disease mouse model: Global atrophy in the absence of white matter microstructural damage

Jessica J. Steventon, Rebecca C. Trueman, Da Ma, Emma Yhnell, Zubeyde Bayram-Weston Orcid Logo, Marc Modat, Jorge Cardoso, Sebastian Ourselin, Mark Lythgoe, Andrew Stewart, Anne E. Rosser, Derek K. Jones

Scientific Reports, Volume: 6, Issue: 1

Swansea University Author: Zubeyde Bayram-Weston Orcid Logo

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DOI (Published version): 10.1038/srep32423

Abstract

Huntington’s disease (HD) is a genetically-determined neurodegenerative disease. Characterising neuropathology in mouse models of HD is commonly restricted to cross-sectional ex vivo analyses, beset by tissue fixation issues. In vivo longitudinal magnetic resonance imaging (MRI) allows for disease p...

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Published in: Scientific Reports
ISSN: 2045-2322
Published: Springer Science and Business Media LLC 2016
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spelling 2022-10-27T15:58:05.6475886 v2 46019 2018-11-21 Longitudinal in vivo MRI in a Huntington’s disease mouse model: Global atrophy in the absence of white matter microstructural damage 93ba509b96e1eacf70cd2afd51361094 0000-0003-4560-8186 Zubeyde Bayram-Weston Zubeyde Bayram-Weston true false 2018-11-21 HEAL Huntington’s disease (HD) is a genetically-determined neurodegenerative disease. Characterising neuropathology in mouse models of HD is commonly restricted to cross-sectional ex vivo analyses, beset by tissue fixation issues. In vivo longitudinal magnetic resonance imaging (MRI) allows for disease progression to be probed non-invasively. In the HdhQ150 mouse model of HD, in vivo MRI was employed at two time points, before and after the onset of motor signs, to assess brain macrostructure and white matter microstructure. Ex vivo MRI, immunohistochemistry, transmission electron microscopy and behavioural testing were also conducted. Global brain atrophy was found in HdhQ150 mice at both time points, with no neuropathological progression across time and a selective sparing of the cerebellum. In contrast, no white matter abnormalities were detected from the MRI images or electron microscopy images alike. The relationship between motor function and MR-based structural measurements was different for the HdhQ150 and wild-type mice, although there was no relationship between motor deficits and histopathology. Widespread neuropathology prior to symptom onset is consistent with patient studies, whereas the absence of white matter abnormalities conflicts with patient data. The myriad reasons for this inconsistency require further attention to improve the translatability from mouse models of disease. Journal Article Scientific Reports 6 1 Springer Science and Business Media LLC 2045-2322 1 9 2016 2016-09-01 10.1038/srep32423 COLLEGE NANME Healthcare Science COLLEGE CODE HEAL Swansea University 2022-10-27T15:58:05.6475886 2018-11-21T13:35:40.6993959 Faculty of Medicine, Health and Life Sciences School of Health and Social Care - Healthcare Science Jessica J. Steventon 1 Rebecca C. Trueman 2 Da Ma 3 Emma Yhnell 4 Zubeyde Bayram-Weston 0000-0003-4560-8186 5 Marc Modat 6 Jorge Cardoso 7 Sebastian Ourselin 8 Mark Lythgoe 9 Andrew Stewart 10 Anne E. Rosser 11 Derek K. Jones 12 0046019-30042019130638.pdf 46019.pdf 2019-04-30T13:06:38.1870000 Output 1441132 application/pdf Version of Record true Released under the terms of a Creative Commons Attribution 4.0 International License (CC-BY). true eng http://creativecommons.org/licenses/by/4.0/
title Longitudinal in vivo MRI in a Huntington’s disease mouse model: Global atrophy in the absence of white matter microstructural damage
spellingShingle Longitudinal in vivo MRI in a Huntington’s disease mouse model: Global atrophy in the absence of white matter microstructural damage
Zubeyde Bayram-Weston
title_short Longitudinal in vivo MRI in a Huntington’s disease mouse model: Global atrophy in the absence of white matter microstructural damage
title_full Longitudinal in vivo MRI in a Huntington’s disease mouse model: Global atrophy in the absence of white matter microstructural damage
title_fullStr Longitudinal in vivo MRI in a Huntington’s disease mouse model: Global atrophy in the absence of white matter microstructural damage
title_full_unstemmed Longitudinal in vivo MRI in a Huntington’s disease mouse model: Global atrophy in the absence of white matter microstructural damage
title_sort Longitudinal in vivo MRI in a Huntington’s disease mouse model: Global atrophy in the absence of white matter microstructural damage
author_id_str_mv 93ba509b96e1eacf70cd2afd51361094
author_id_fullname_str_mv 93ba509b96e1eacf70cd2afd51361094_***_Zubeyde Bayram-Weston
author Zubeyde Bayram-Weston
author2 Jessica J. Steventon
Rebecca C. Trueman
Da Ma
Emma Yhnell
Zubeyde Bayram-Weston
Marc Modat
Jorge Cardoso
Sebastian Ourselin
Mark Lythgoe
Andrew Stewart
Anne E. Rosser
Derek K. Jones
format Journal article
container_title Scientific Reports
container_volume 6
container_issue 1
publishDate 2016
institution Swansea University
issn 2045-2322
doi_str_mv 10.1038/srep32423
publisher Springer Science and Business Media LLC
college_str Faculty of Medicine, Health and Life Sciences
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hierarchy_top_id facultyofmedicinehealthandlifesciences
hierarchy_top_title Faculty of Medicine, Health and Life Sciences
hierarchy_parent_id facultyofmedicinehealthandlifesciences
hierarchy_parent_title Faculty of Medicine, Health and Life Sciences
department_str School of Health and Social Care - Healthcare Science{{{_:::_}}}Faculty of Medicine, Health and Life Sciences{{{_:::_}}}School of Health and Social Care - Healthcare Science
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description Huntington’s disease (HD) is a genetically-determined neurodegenerative disease. Characterising neuropathology in mouse models of HD is commonly restricted to cross-sectional ex vivo analyses, beset by tissue fixation issues. In vivo longitudinal magnetic resonance imaging (MRI) allows for disease progression to be probed non-invasively. In the HdhQ150 mouse model of HD, in vivo MRI was employed at two time points, before and after the onset of motor signs, to assess brain macrostructure and white matter microstructure. Ex vivo MRI, immunohistochemistry, transmission electron microscopy and behavioural testing were also conducted. Global brain atrophy was found in HdhQ150 mice at both time points, with no neuropathological progression across time and a selective sparing of the cerebellum. In contrast, no white matter abnormalities were detected from the MRI images or electron microscopy images alike. The relationship between motor function and MR-based structural measurements was different for the HdhQ150 and wild-type mice, although there was no relationship between motor deficits and histopathology. Widespread neuropathology prior to symptom onset is consistent with patient studies, whereas the absence of white matter abnormalities conflicts with patient data. The myriad reasons for this inconsistency require further attention to improve the translatability from mouse models of disease.
published_date 2016-09-01T03:57:42Z
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