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Time estimation and arousal responses in dopa-responsive dystonia
Dopa-responsive dystonia (DRD) is caused by an impaired dopamine biosynthesis due to a GTP-cyclohydrolase-1 (GCH1) deficiency, resulting in a combination of dystonia and parkinsonism. However, the effect of GCH1 mutations and levodopa treatment on motor control beyond simple movements, such as timin...
Autores principales: | , , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Nature Publishing Group UK
2022
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9395389/ https://www.ncbi.nlm.nih.gov/pubmed/35995805 http://dx.doi.org/10.1038/s41598-022-17545-w |
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author | Becker, Leonie F. Tunc, Sinem Murphy, Peter Bäumer, Tobias Weissbach, Anne Pauly, Martje G. Al-Shorafat, Duha M. Saranza, Gerard Lang, Anthony E. Beste, Christian Donner, Tobias H. Verrel, Julius Münchau, Alexander |
author_facet | Becker, Leonie F. Tunc, Sinem Murphy, Peter Bäumer, Tobias Weissbach, Anne Pauly, Martje G. Al-Shorafat, Duha M. Saranza, Gerard Lang, Anthony E. Beste, Christian Donner, Tobias H. Verrel, Julius Münchau, Alexander |
author_sort | Becker, Leonie F. |
collection | PubMed |
description | Dopa-responsive dystonia (DRD) is caused by an impaired dopamine biosynthesis due to a GTP-cyclohydrolase-1 (GCH1) deficiency, resulting in a combination of dystonia and parkinsonism. However, the effect of GCH1 mutations and levodopa treatment on motor control beyond simple movements, such as timing, action preparation and feedback processing, have not been investigated so far. In an active time estimation task with trial-by-trial feedback, participants indicated a target interval (1200 ms) by a motor response. We compared 12 patients tested (in fixed order) under their current levodopa medication ("ON") and after levodopa withdrawal ("OFF") to matched healthy controls (HC), measured twice to control for repetition effects. We assessed time estimation accuracy, trial-to-trial adjustment, as well as task- and feedback-related pupil-linked arousal responses. Patients showed comparable time estimation accuracy ON medication as HC but reduced performance OFF medication. Task-related pupil responses showed the reverse pattern. Trial-to-trial adjustments of response times were reduced in DRD, particularly OFF medication. Our results indicate differential alterations of time estimation accuracy and task-related arousal dynamics in DRD patients as a function of dopaminergic medication state. A medication-independent alteration of task repetition effects in DRD cannot be ruled out with certainty but is discussed as less likely. |
format | Online Article Text |
id | pubmed-9395389 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | Nature Publishing Group UK |
record_format | MEDLINE/PubMed |
spelling | pubmed-93953892022-08-24 Time estimation and arousal responses in dopa-responsive dystonia Becker, Leonie F. Tunc, Sinem Murphy, Peter Bäumer, Tobias Weissbach, Anne Pauly, Martje G. Al-Shorafat, Duha M. Saranza, Gerard Lang, Anthony E. Beste, Christian Donner, Tobias H. Verrel, Julius Münchau, Alexander Sci Rep Article Dopa-responsive dystonia (DRD) is caused by an impaired dopamine biosynthesis due to a GTP-cyclohydrolase-1 (GCH1) deficiency, resulting in a combination of dystonia and parkinsonism. However, the effect of GCH1 mutations and levodopa treatment on motor control beyond simple movements, such as timing, action preparation and feedback processing, have not been investigated so far. In an active time estimation task with trial-by-trial feedback, participants indicated a target interval (1200 ms) by a motor response. We compared 12 patients tested (in fixed order) under their current levodopa medication ("ON") and after levodopa withdrawal ("OFF") to matched healthy controls (HC), measured twice to control for repetition effects. We assessed time estimation accuracy, trial-to-trial adjustment, as well as task- and feedback-related pupil-linked arousal responses. Patients showed comparable time estimation accuracy ON medication as HC but reduced performance OFF medication. Task-related pupil responses showed the reverse pattern. Trial-to-trial adjustments of response times were reduced in DRD, particularly OFF medication. Our results indicate differential alterations of time estimation accuracy and task-related arousal dynamics in DRD patients as a function of dopaminergic medication state. A medication-independent alteration of task repetition effects in DRD cannot be ruled out with certainty but is discussed as less likely. Nature Publishing Group UK 2022-08-22 /pmc/articles/PMC9395389/ /pubmed/35995805 http://dx.doi.org/10.1038/s41598-022-17545-w Text en © The Author(s) 2022 https://creativecommons.org/licenses/by/4.0/Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) . |
spellingShingle | Article Becker, Leonie F. Tunc, Sinem Murphy, Peter Bäumer, Tobias Weissbach, Anne Pauly, Martje G. Al-Shorafat, Duha M. Saranza, Gerard Lang, Anthony E. Beste, Christian Donner, Tobias H. Verrel, Julius Münchau, Alexander Time estimation and arousal responses in dopa-responsive dystonia |
title | Time estimation and arousal responses in dopa-responsive dystonia |
title_full | Time estimation and arousal responses in dopa-responsive dystonia |
title_fullStr | Time estimation and arousal responses in dopa-responsive dystonia |
title_full_unstemmed | Time estimation and arousal responses in dopa-responsive dystonia |
title_short | Time estimation and arousal responses in dopa-responsive dystonia |
title_sort | time estimation and arousal responses in dopa-responsive dystonia |
topic | Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9395389/ https://www.ncbi.nlm.nih.gov/pubmed/35995805 http://dx.doi.org/10.1038/s41598-022-17545-w |
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