{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,11]],"date-time":"2026-09-11T01:02:48Z","timestamp":1789088568069,"version":"build-2803163510"},"reference-count":36,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2023,12,20]],"date-time":"2023-12-20T00:00:00Z","timestamp":1703030400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000071","name":"National Institute of Child Health and Human Development","doi-asserted-by":"publisher","award":["HD036071"],"award-info":[{"award-number":["HD036071"]}],"id":[{"id":"10.13039\/100000071","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000071","name":"National Institute of Child Health and Human Development","doi-asserted-by":"publisher","award":["P50HD103526"],"award-info":[{"award-number":["P50HD103526"]}],"id":[{"id":"10.13039\/100000071","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["movementdisorders.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Movement Disorders"],"published-print":{"date-parts":[[2024,3]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Men with fragile X\u2010associated tremor\/ataxia syndrome (FXTAS) often develop executive dysfunction, characterized by disinhibition, frontal dyscontrol of movement, and working memory and attention changes. Although cross\u2010sectional studies have suggested that earlier executive function changes may precede FXTAS, the lack of longitudinal studies has made it difficult to address this hypothesis.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>To determine whether executive function deterioration experienced by premutation carriers (PC) in daily life precedes and predicts FXTAS.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>\n                      This study included 66\n                      <jats:italic>FMR1<\/jats:italic>\n                      PC ranging from 40 to 78\u2009years (mean,\u200959.5) and 31 well\u2010matched healthy controls (HC) ages 40 to 75 (mean, 57.7) at baseline. Eighty\u2010four participants returned for 2 to 5 follow up visits over a duration of 1 to 9\u2009years (mean,\u20094.6); 28 of the PC developed FXTAS. The Behavior Rating Inventory of Executive Function\u2010Adult Version (BRIEF\u2010A) was completed by participants and their spouses\/partners at each visit.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Longitudinal mixed model regression analyses showed a greater decline with age in PC compared to HC on the Metacognition Index (MI; self\u2010initiation, working memory, organization, task monitoring). Conversion to FXTAS was associated with worsening MI and Behavioral Regulation Index (BRI; inhibition, flexibility, emotion modulation). For spouse\/partner report, FXTAS conversion was associated with worsening MI. Finally, increased self\u2010report executive function problems at baseline significantly predicted later development of FXTAS.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>\n                      Executive function changes experienced by male PC represent a prodrome of the later movement disorder. \u00a9 2023 The Authors.\n                      <jats:italic>Movement Disorders<\/jats:italic>\n                      published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/mds.29695","type":"journal-article","created":{"date-parts":[[2023,12,21]],"date-time":"2023-12-21T00:59:53Z","timestamp":1703120393000},"page":"519-525","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":12,"title":["<scp>\n                      <i>FMR1<\/i>\n                    <\/scp>\n                    Carriers Report Executive Function Changes Prior to\n                    <scp>Fragile X\u2010Associated Tremor\/Ataxia Syndrome<\/scp>\n                    : A Longitudinal Study"],"prefix":"10.1002","volume":"39","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-3460-9805","authenticated-orcid":false,"given":"David","family":"Hessl","sequence":"first","affiliation":[{"name":"MIND Institute, University of California Davis Health  Sacramento California USA"},{"name":"Department of Psychiatry and Behavioral Sciences University of California Davis School of Medicine  Sacramento California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8445-2167","authenticated-orcid":false,"given":"Karina","family":"Mandujano Rojas","sequence":"additional","affiliation":[{"name":"MIND Institute, University of California Davis Health  Sacramento California 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