Impaired saccadic adaptation in DYT11 dystonia - Archive ouverte HAL
Article Dans Une Revue Journal of Neurology, Neurosurgery and Psychiatry Année : 2011

Impaired saccadic adaptation in DYT11 dystonia

Résumé

Background: Recent neuroimaging studies point to a possible pathophysiological role of cerebellar dysfunction in dystonia. The authors investigated the association between sensorimotor adaptation, cerebellar dysfunction and the myoclonus-dystonia phenotype. Methods: The authors prospectively analysed reactive saccade adaptation in a genetically homogeneous group of 14 patients with DYT11 dystonia owing to a mutation of the SGCE gene. The authors used a backward reactive saccade adaptation task, a well-characterised experimental oculomotor paradigm involving the cerebellum. The principle of this paradigm is to simulate a spatial error in saccade generation by systematically shifting a visual target during saccade execution. Repetition of this systematic error induces a gradual decrease in the initial saccade amplitude, reflecting an adaptive phenomenon. Results: Saccade adaptation was significantly lower in the DYT11 patients than in healthy controls (mean value: 8.9%±4.5% vs 21.6%±4.5%; p=8.3×10(-6)). The time course of adaptation also differed between the patients and controls (p=0.002), reflecting the slower saccadic adaptation in the patients. Conclusions: This study provides the first neurophysiological evidence of cerebellar dysfunction in DYT11 dystonia and supports a role of cerebellar dysfunction in the myoclonus-dystonia phenotype.

Dates et versions

hal-02196716 , version 1 (03-07-2023)

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Citer

Cécile Hubsch, Marie Vidailhet, Sophie Rivaud-Péchoux, Pierre Pouget, Vanessa Brochard, et al.. Impaired saccadic adaptation in DYT11 dystonia. Journal of Neurology, Neurosurgery and Psychiatry, 2011, 82 (10), pp.1103-1106. ⟨10.1136/jnnp.2010.232793⟩. ⟨hal-02196716⟩
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