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Macro- and micro-structural insights into primary dystonia: a UK Biobank study

Claire L. MacIver Orcid Logo, Grace Bailey Orcid Logo, Pedro Luque Laguna, Megan E. Wadon, Ann-Kathrin Schalkamp, Cynthia Sandor, Derek K. Jones, Chantal M. W. Tax, Kathryn J. Peall

Journal of Neurology, Volume: 271, Issue: 3, Pages: 1416 - 1427

Swansea University Author: Grace Bailey Orcid Logo

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DOI (Published version): 10.1007/s00415-023-12086-2

Abstract

BackgroundDystonia is a hyperkinetic movement disorder with key motor network dysfunction implicated in pathophysiology. The UK Biobank encompasses > 500,000 participants, of whom 42,565 underwent brain MRI scanning. This study applied an optimized pre-processing pipeline, aimed at better account...

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Published in: Journal of Neurology
ISSN: 0340-5354 1432-1459
Published: Springer Science and Business Media LLC 2024
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URI: https://cronfa.swan.ac.uk/Record/cronfa66534
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This study applied an optimized pre-processing pipeline, aimed at better accounting for artifact and improving data reliability, to assess for grey and white matter structural MRI changes between individuals diagnosed with primary dystonia and an unaffected control cohort.MethodsIndividuals with dystonia (n = 76) were identified from the UK Biobank using published algorithms, alongside an age- and sex-matched unaffected control cohort (n = 311). Grey matter morphometric and diffusion measures were assessed, together with white matter diffusion tensor and diffusion kurtosis metrics using tractography and tractometry. Post-hoc Neurite Orientation and Density Distribution Imaging (NODDI) was also undertaken for tracts in which significant differences were observed.ResultsGrey matter tremor-specific striatal differences were observed, with higher radial kurtosis. Tractography identified no white matter differences, however segmental tractometry identified localised differences, particularly in the superior cerebellar peduncles and anterior thalamic radiations, including higher fractional anisotropy and lower orientation distribution index in dystonia, compared to controls. Additional tremor-specific changes included lower neurite density index in the anterior thalamic radiations.ConclusionsAnalysis of imaging data from one of the largest dystonia cohorts to date demonstrates microstructural differences in cerebellar and thalamic white matter connections, with architectural differences such as less orientation dispersion potentially being a component of the morphological structural changes implicated in dystonia. 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KJP is funded by an MRC Clinician-Scientist Fellowship &amp; Transition Award (MR/P008593/1, MR/V036084/1). CMWT is supported by a Sir Henry Wellcome Fellowship (215,944/Z/19/Z) and a Veni grant from the Dutch Research Council (NWO) (17,331). 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spelling v2 66534 2024-05-29 Macro- and micro-structural insights into primary dystonia: a UK Biobank study 1e09a407fca9e8047e7738b18d381130 0000-0003-4646-3134 Grace Bailey Grace Bailey true false 2024-05-29 MEDS BackgroundDystonia is a hyperkinetic movement disorder with key motor network dysfunction implicated in pathophysiology. The UK Biobank encompasses > 500,000 participants, of whom 42,565 underwent brain MRI scanning. This study applied an optimized pre-processing pipeline, aimed at better accounting for artifact and improving data reliability, to assess for grey and white matter structural MRI changes between individuals diagnosed with primary dystonia and an unaffected control cohort.MethodsIndividuals with dystonia (n = 76) were identified from the UK Biobank using published algorithms, alongside an age- and sex-matched unaffected control cohort (n = 311). Grey matter morphometric and diffusion measures were assessed, together with white matter diffusion tensor and diffusion kurtosis metrics using tractography and tractometry. Post-hoc Neurite Orientation and Density Distribution Imaging (NODDI) was also undertaken for tracts in which significant differences were observed.ResultsGrey matter tremor-specific striatal differences were observed, with higher radial kurtosis. Tractography identified no white matter differences, however segmental tractometry identified localised differences, particularly in the superior cerebellar peduncles and anterior thalamic radiations, including higher fractional anisotropy and lower orientation distribution index in dystonia, compared to controls. Additional tremor-specific changes included lower neurite density index in the anterior thalamic radiations.ConclusionsAnalysis of imaging data from one of the largest dystonia cohorts to date demonstrates microstructural differences in cerebellar and thalamic white matter connections, with architectural differences such as less orientation dispersion potentially being a component of the morphological structural changes implicated in dystonia. Distinct tremor-related imaging features are also implicated in both grey and white matter. Journal Article Journal of Neurology 271 3 1416 1427 Springer Science and Business Media LLC 0340-5354 1432-1459 Dystonia; MRI; Diffusion MRI; Structural MRI 1 3 2024 2024-03-01 10.1007/s00415-023-12086-2 COLLEGE NANME Medical School COLLEGE CODE MEDS Swansea University Another institution paid the OA fee This work was supported by an ABN/Guarantors of Brain Clinical Research Training Fellowship (520,286) and a Wellcome Trust Translation of Concept Scheme (Institutional Translational Partnership Award) (520,958). KJP is funded by an MRC Clinician-Scientist Fellowship & Transition Award (MR/P008593/1, MR/V036084/1). CMWT is supported by a Sir Henry Wellcome Fellowship (215,944/Z/19/Z) and a Veni grant from the Dutch Research Council (NWO) (17,331). DKJ is supported by Wellcome Trust grants (096646/Z/11/Z and 104,943/Z/14/Z). 2024-06-19T12:50:57.4548870 2024-05-29T20:05:18.3167171 Faculty of Medicine, Health and Life Sciences Swansea University Medical School - Health Data Science Claire L. MacIver 0000-0002-5040-3487 1 Grace Bailey 0000-0003-4646-3134 2 Pedro Luque Laguna 3 Megan E. Wadon 4 Ann-Kathrin Schalkamp 5 Cynthia Sandor 6 Derek K. Jones 7 Chantal M. W. Tax 8 Kathryn J. Peall 9 66534__30574__9ae08b3c0eb644cb95d9a5cccc3874a8.pdf 65534.pdf 2024-06-07T15:18:50.9250166 Output 3960317 application/pdf Version of Record true This article is licensed under a Creative Commons Attribution 4.0 International License. true eng https://creativecommons.org/licenses/by/4.0/
title Macro- and micro-structural insights into primary dystonia: a UK Biobank study
spellingShingle Macro- and micro-structural insights into primary dystonia: a UK Biobank study
Grace Bailey
title_short Macro- and micro-structural insights into primary dystonia: a UK Biobank study
title_full Macro- and micro-structural insights into primary dystonia: a UK Biobank study
title_fullStr Macro- and micro-structural insights into primary dystonia: a UK Biobank study
title_full_unstemmed Macro- and micro-structural insights into primary dystonia: a UK Biobank study
title_sort Macro- and micro-structural insights into primary dystonia: a UK Biobank study
author_id_str_mv 1e09a407fca9e8047e7738b18d381130
author_id_fullname_str_mv 1e09a407fca9e8047e7738b18d381130_***_Grace Bailey
author Grace Bailey
author2 Claire L. MacIver
Grace Bailey
Pedro Luque Laguna
Megan E. Wadon
Ann-Kathrin Schalkamp
Cynthia Sandor
Derek K. Jones
Chantal M. W. Tax
Kathryn J. Peall
format Journal article
container_title Journal of Neurology
container_volume 271
container_issue 3
container_start_page 1416
publishDate 2024
institution Swansea University
issn 0340-5354
1432-1459
doi_str_mv 10.1007/s00415-023-12086-2
publisher Springer Science and Business Media LLC
college_str Faculty of Medicine, Health and Life Sciences
hierarchytype
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 Swansea University Medical School - Health Data Science{{{_:::_}}}Faculty of Medicine, Health and Life Sciences{{{_:::_}}}Swansea University Medical School - Health Data Science
document_store_str 1
active_str 0
description BackgroundDystonia is a hyperkinetic movement disorder with key motor network dysfunction implicated in pathophysiology. The UK Biobank encompasses > 500,000 participants, of whom 42,565 underwent brain MRI scanning. This study applied an optimized pre-processing pipeline, aimed at better accounting for artifact and improving data reliability, to assess for grey and white matter structural MRI changes between individuals diagnosed with primary dystonia and an unaffected control cohort.MethodsIndividuals with dystonia (n = 76) were identified from the UK Biobank using published algorithms, alongside an age- and sex-matched unaffected control cohort (n = 311). Grey matter morphometric and diffusion measures were assessed, together with white matter diffusion tensor and diffusion kurtosis metrics using tractography and tractometry. Post-hoc Neurite Orientation and Density Distribution Imaging (NODDI) was also undertaken for tracts in which significant differences were observed.ResultsGrey matter tremor-specific striatal differences were observed, with higher radial kurtosis. Tractography identified no white matter differences, however segmental tractometry identified localised differences, particularly in the superior cerebellar peduncles and anterior thalamic radiations, including higher fractional anisotropy and lower orientation distribution index in dystonia, compared to controls. Additional tremor-specific changes included lower neurite density index in the anterior thalamic radiations.ConclusionsAnalysis of imaging data from one of the largest dystonia cohorts to date demonstrates microstructural differences in cerebellar and thalamic white matter connections, with architectural differences such as less orientation dispersion potentially being a component of the morphological structural changes implicated in dystonia. Distinct tremor-related imaging features are also implicated in both grey and white matter.
published_date 2024-03-01T12:50:56Z
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