Prosaccade and Antisaccade Behavior in Fragile X-Associated Tremor/Ataxia Syndrome Progression

Yingratana A McLennan; Matthew W Mosconi; Forrest J McKenzie; Jessica Famula; Bennet Krawchuk; Kyoungmi Kim; Courtney J Clark; David Hessl; Susan M Rivera; Tony J Simon; Flora Tassone; Randi J Hagerman

doi:10.1002/mdc3.13449

Prosaccade and Antisaccade Behavior in Fragile X-Associated Tremor/Ataxia Syndrome Progression

Mov Disord Clin Pract. 2022 Apr 28;9(4):473-478. doi: 10.1002/mdc3.13449. eCollection 2022 May.

Authors

Yingratana A McLennan^{1

2}, Matthew W Mosconi³, Forrest J McKenzie⁴, Jessica Famula^{1

5}, Bennet Krawchuk⁴, Kyoungmi Kim⁶, Courtney J Clark^{1

2}, David Hessl^{1

4}, Susan M Rivera^{1

6}, Tony J Simon^{1

4}, Flora Tassone^{1

7}, Randi J Hagerman^{1

2}

Affiliations

¹ The MIND Institute University of California Davis Medical Center Sacramento California USA.
² Department of Pediatrics University of California Davis Medical Center Sacramento California USA.
³ Life Span Institute, Kansas Center for Autism Research and Training, and Clinical Child Psychology Program University of Kansas Lawrence Kansas USA.
⁴ University of California Davis School of Medicine Sacramento California USA.
⁵ Department of Psychiatry and Behavioral Sciences University of California Davis Medical Center Sacramento California USA.
⁶ Department of Psychology University of California Davis California USA.
⁷ Department of Biochemistry and Molecular Medicine University of California Davis School of Medicine Sacramento California USA.

Abstract

Background: Quantitative measurement of eye movements can reveal subtle progression in neurodegenerative diseases.

Objective: To determine if quantitative measurements of eye movements may reveal subtle progression of fragile X-associated tremor and ataxia (FXTAS).

Methods: Prosaccade (PS) and antisaccade (AS) behavior was analyzed in 25 controls, 57 non-FXTAS carriers, and 46 carriers with FXTAS.

Results: Symptomatic individuals with FXTAS had longer AS latencies, increased rates of AS errors, and increased AS dysmetria relative to non-FXTAS carriers and controls. These deficits, along with PS latency and velocity, were greater in advanced FXTAS stages.

Conclusion: AS deficits differentiated FXTAS from non-FXTAS premutation carriers implicating top-down control and frontostriatal deterioration. However, the absence of group differences between non-FXTAS carriers and controls in AS and PS markers suggests saccade performance may not be a sensitive enough measure for detecting conversion to FXTAS, but instead more helpful as translational biomarkers of FXTAS progression.

Keywords: FXTAS; antisaccade; eye movements; inhibitory deficits.; prosaccade.

Abstract

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