Time estimation and arousal responses in dopa-responsive dystonia.

Autor: Becker LF; Institute of Systems Motor Science, Center of Brain, Behavior and Metabolism, Universität zu Lübeck, Ratzeburger Allee 160, 23538, Lübeck, Germany., Tunc S; Institute of Systems Motor Science, Center of Brain, Behavior and Metabolism, Universität zu Lübeck, Ratzeburger Allee 160, 23538, Lübeck, Germany.; Department of Neurology, University Hospital Schleswig Holstein, Lübeck, Germany., Murphy P; Section Computational Cognitive Neuroscience, Department of Neurophysiology and Pathophysiology, University Medical Center Hamburg-Eppendorf, Hamburg, Germany.; School of Psychology, Trinity College Institute of Neuroscience, Trinity College Dublin, Dublin, Ireland.; Bernstein Center for Computational Neuroscience Berlin, Berlin, Germany., Bäumer T; Institute of Systems Motor Science, Center of Brain, Behavior and Metabolism, Universität zu Lübeck, Ratzeburger Allee 160, 23538, Lübeck, Germany., Weissbach A; Institute of Systems Motor Science, Center of Brain, Behavior and Metabolism, Universität zu Lübeck, Ratzeburger Allee 160, 23538, Lübeck, Germany.; Institute of Neurogenetics, Universität zu Lübeck, Lübeck, Germany., Pauly MG; Institute of Systems Motor Science, Center of Brain, Behavior and Metabolism, Universität zu Lübeck, Ratzeburger Allee 160, 23538, Lübeck, Germany.; Department of Neurology, University Hospital Schleswig Holstein, Lübeck, Germany.; Institute of Neurogenetics, Universität zu Lübeck, Lübeck, Germany., Al-Shorafat DM; Edmond J. Safra Program in Parkinson's Disease, Morton and Gloria Shulman Movement Disorders Clinic, Toronto Western Hospital, Toronto, ON, Canada.; Neuroscience Department, Jordan University of Science and Technology, Irbid, Jordan., Saranza G; Edmond J. Safra Program in Parkinson's Disease, Morton and Gloria Shulman Movement Disorders Clinic, Toronto Western Hospital, Toronto, ON, Canada.; Section of Neurology, Department of Medicine, Chong Hua Hospital, Cebu City, Philippines., Lang AE; Edmond J. Safra Program in Parkinson's Disease, Morton and Gloria Shulman Movement Disorders Clinic, Toronto Western Hospital, Toronto, ON, Canada., Beste C; Cognitive Neurophysiology, Department of Child and Adolescent Psychiatry, Faculty of Medicine, TU Dresden, Dresden, Germany., Donner TH; Section Computational Cognitive Neuroscience, Department of Neurophysiology and Pathophysiology, University Medical Center Hamburg-Eppendorf, Hamburg, Germany., Verrel J; Institute of Systems Motor Science, Center of Brain, Behavior and Metabolism, Universität zu Lübeck, Ratzeburger Allee 160, 23538, Lübeck, Germany., Münchau A; Institute of Systems Motor Science, Center of Brain, Behavior and Metabolism, Universität zu Lübeck, Ratzeburger Allee 160, 23538, Lübeck, Germany. alexander.muenchau@neuro.uni-luebeck.de.
Jazyk: angličtina
Zdroj: Scientific reports [Sci Rep] 2022 Aug 22; Vol. 12 (1), pp. 14279. Date of Electronic Publication: 2022 Aug 22.
DOI: 10.1038/s41598-022-17545-w
Abstrakt: 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.
(© 2022. The Author(s).)
Databáze: MEDLINE
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