Sensorimotor and inhibitory control in aging FMR1 premutation carriers
Aging FMR1 premutation carriers are at risk of developing neurodegenerative disorders, including fragile X-associated tremor/ataxia syndrome (FXTAS), and there is a need to identify biomarkers that can aid in identification and treatment of these disorders. While FXTAS is more common in males than f...
| المؤلفون الرئيسيون: | , , , , , , |
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| التنسيق: | مقال |
| اللغة: | English |
| منشور في: |
Frontiers Media S.A.
2023-11-01
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| سلاسل: | Frontiers in Human Neuroscience |
| الموضوعات: | |
| الوصول للمادة أونلاين: | https://www.frontiersin.org/articles/10.3389/fnhum.2023.1271158/full |
| _version_ | 1827759354768523264 |
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| author | Heather Fielding-Gebhardt Shannon E. Kelly Kathryn E. Unruh Kathryn E. Unruh Lauren M. Schmitt Lauren M. Schmitt Stormi L. Pulver Pravin Khemani Matthew W. Mosconi Matthew W. Mosconi Matthew W. Mosconi |
| author_facet | Heather Fielding-Gebhardt Shannon E. Kelly Kathryn E. Unruh Kathryn E. Unruh Lauren M. Schmitt Lauren M. Schmitt Stormi L. Pulver Pravin Khemani Matthew W. Mosconi Matthew W. Mosconi Matthew W. Mosconi |
| author_sort | Heather Fielding-Gebhardt |
| collection | DOAJ |
| description | Aging FMR1 premutation carriers are at risk of developing neurodegenerative disorders, including fragile X-associated tremor/ataxia syndrome (FXTAS), and there is a need to identify biomarkers that can aid in identification and treatment of these disorders. While FXTAS is more common in males than females, females can develop the disease, and some evidence suggests that patterns of impairment may differ across sexes. Few studies include females with symptoms of FXTAS, and as a result, little information is available on key phenotypes for tracking disease risk and progression in female premutation carriers. Our aim was to examine quantitative motor and cognitive traits in aging premutation carriers. We administered oculomotor tests of visually guided/reactive saccades (motor) and antisaccades (cognitive control) in 22 premutation carriers (73% female) and 32 age- and sex-matched healthy controls. Neither reactive saccade latency nor accuracy differed between groups. FMR1 premutation carriers showed increased antisaccade latencies relative to controls, both when considering males and females together and when analyzing females separately. Reduced saccade accuracy and increased antisaccade latency each were associated with more severe clinically rated neuromotor impairments. Findings indicate that together male and female premutation carriers show a reduced ability to rapidly exert volitional control over prepotent responses and that quantitative differences in oculomotor behavior, including control of visually guided and antisaccades, may track with FXTAS – related degeneration in male and female premutation carriers. |
| first_indexed | 2024-03-11T09:22:12Z |
| format | Article |
| id | doaj.art-73a9bf28742f444cbf407b716395348c |
| institution | Directory Open Access Journal |
| issn | 1662-5161 |
| language | English |
| last_indexed | 2024-03-11T09:22:12Z |
| publishDate | 2023-11-01 |
| publisher | Frontiers Media S.A. |
| record_format | Article |
| series | Frontiers in Human Neuroscience |
| spelling | doaj.art-73a9bf28742f444cbf407b716395348c2023-11-16T18:13:19ZengFrontiers Media S.A.Frontiers in Human Neuroscience1662-51612023-11-011710.3389/fnhum.2023.12711581271158Sensorimotor and inhibitory control in aging FMR1 premutation carriersHeather Fielding-Gebhardt0Shannon E. Kelly1Kathryn E. Unruh2Kathryn E. Unruh3Lauren M. Schmitt4Lauren M. Schmitt5Stormi L. Pulver6Pravin Khemani7Matthew W. Mosconi8Matthew W. Mosconi9Matthew W. Mosconi10Life Span Institute, University of Kansas, Lawrence, KS, United StatesScholars Strategy Network, Boston, MA, United StatesLife Span Institute, University of Kansas, Lawrence, KS, United StatesKansas Center for Autism Research and Training (K-CART), University of Kansas, Lawrence, KS, United StatesDivision of Behavioral Medicine and Clinical Psychology, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH, United StatesDepartment of Pediatrics, University of Cincinnati College of Medicine, Cincinnati, OH, United StatesDivision of Autism and Related Disorders, Emory University School of Medicine, Atlanta, GA, United StatesMovement Disorders Program, Swedish Neuroscience Institute, Seattle, WA, United StatesLife Span Institute, University of Kansas, Lawrence, KS, United StatesKansas Center for Autism Research and Training (K-CART), University of Kansas, Lawrence, KS, United StatesClinical Child Psychology Program, University of Kansas, Lawrence, KS, United StatesAging FMR1 premutation carriers are at risk of developing neurodegenerative disorders, including fragile X-associated tremor/ataxia syndrome (FXTAS), and there is a need to identify biomarkers that can aid in identification and treatment of these disorders. While FXTAS is more common in males than females, females can develop the disease, and some evidence suggests that patterns of impairment may differ across sexes. Few studies include females with symptoms of FXTAS, and as a result, little information is available on key phenotypes for tracking disease risk and progression in female premutation carriers. Our aim was to examine quantitative motor and cognitive traits in aging premutation carriers. We administered oculomotor tests of visually guided/reactive saccades (motor) and antisaccades (cognitive control) in 22 premutation carriers (73% female) and 32 age- and sex-matched healthy controls. Neither reactive saccade latency nor accuracy differed between groups. FMR1 premutation carriers showed increased antisaccade latencies relative to controls, both when considering males and females together and when analyzing females separately. Reduced saccade accuracy and increased antisaccade latency each were associated with more severe clinically rated neuromotor impairments. Findings indicate that together male and female premutation carriers show a reduced ability to rapidly exert volitional control over prepotent responses and that quantitative differences in oculomotor behavior, including control of visually guided and antisaccades, may track with FXTAS – related degeneration in male and female premutation carriers.https://www.frontiersin.org/articles/10.3389/fnhum.2023.1271158/fullFXTASFMR1 premutationantisaccadeinhibitory controleye movements |
| spellingShingle | Heather Fielding-Gebhardt Shannon E. Kelly Kathryn E. Unruh Kathryn E. Unruh Lauren M. Schmitt Lauren M. Schmitt Stormi L. Pulver Pravin Khemani Matthew W. Mosconi Matthew W. Mosconi Matthew W. Mosconi Sensorimotor and inhibitory control in aging FMR1 premutation carriers Frontiers in Human Neuroscience FXTAS FMR1 premutation antisaccade inhibitory control eye movements |
| title | Sensorimotor and inhibitory control in aging FMR1 premutation carriers |
| title_full | Sensorimotor and inhibitory control in aging FMR1 premutation carriers |
| title_fullStr | Sensorimotor and inhibitory control in aging FMR1 premutation carriers |
| title_full_unstemmed | Sensorimotor and inhibitory control in aging FMR1 premutation carriers |
| title_short | Sensorimotor and inhibitory control in aging FMR1 premutation carriers |
| title_sort | sensorimotor and inhibitory control in aging fmr1 premutation carriers |
| topic | FXTAS FMR1 premutation antisaccade inhibitory control eye movements |
| url | https://www.frontiersin.org/articles/10.3389/fnhum.2023.1271158/full |
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