Time estimation and arousal responses in dopa-responsive dystonia

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

Abstract

AbstractDopa-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.

Funder

Deutsche Forschungsgemeinschaft

Else Kröner-Fresenius-Stiftung

Universität zu Lübeck

Publisher

Springer Science and Business Media LLC

Subject

Multidisciplinary

Cited by 2 articles. 订阅此论文施引文献 订阅此论文施引文献,注册后可以免费订阅5篇论文的施引文献,订阅后可以查看论文全部施引文献

1. Exploring the interplay between addiction and time perception: A systematic review and meta-analysis;Progress in Neuro-Psychopharmacology and Biological Psychiatry;2024-12

2. Bradykinesia and dystonia;Dystonia;2023-08-08

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