Ageing and cognition in men with fragile X syndrome
Background Fragile X syndrome (FXS) is the most common inherited cause of intellectual disability. The aim of our longitudinal study was to describe ageing‐related cognitive changes in men with FXS. Method A neuropsychologist determined the raw scores (RSs) of 19 men with FXS twice with the Leiter I...
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Veröffentlicht in: | Journal of applied research in intellectual disabilities 2020-09, Vol.33 (5), p.1113-1118 |
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creator | Sauna‐aho, Oili Bjelogrlic‐Laakso, Nina Rautava, Päivi Arvio, Maria |
description | Background
Fragile X syndrome (FXS) is the most common inherited cause of intellectual disability. The aim of our longitudinal study was to describe ageing‐related cognitive changes in men with FXS.
Method
A neuropsychologist determined the raw scores (RSs) of 19 men with FXS twice with the Leiter International Performance Scale at an average interval of 22 years. The ages of the participants at baseline ranged from 16 to 50 (mean 27) years.
Results
At follow‐up, the RSs improved in two men, remained the same in two men and declined in 15 men. Overall, the RS of the study group deteriorated by an average 4 points in RSs (p |
doi_str_mv | 10.1111/jar.12733 |
format | Article |
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Fragile X syndrome (FXS) is the most common inherited cause of intellectual disability. The aim of our longitudinal study was to describe ageing‐related cognitive changes in men with FXS.
Method
A neuropsychologist determined the raw scores (RSs) of 19 men with FXS twice with the Leiter International Performance Scale at an average interval of 22 years. The ages of the participants at baseline ranged from 16 to 50 (mean 27) years.
Results
At follow‐up, the RSs improved in two men, remained the same in two men and declined in 15 men. Overall, the RS of the study group deteriorated by an average 4 points in RSs (p < .001).
Conclusion
Cognitive ageing in men with FXS started earlier than that in men in the general population; in many cases, cognitive ageing in men with FXS began before middle age, usually without any medical or other underlying cause.</description><identifier>ISSN: 1360-2322</identifier><identifier>EISSN: 1468-3148</identifier><identifier>DOI: 10.1111/jar.12733</identifier><identifier>PMID: 32281257</identifier><language>eng</language><publisher>England: Wiley</publisher><subject>Adolescents ; Adults ; ageing ; Aging ; Aging (Individuals) ; Cognitive Ability ; cognitive development ; fragile X syndrome ; Genetic disorders ; Intellectual disabilities ; Intellectual Disability ; Longitudinal Studies ; Males ; Mens health ; Neuropsychology ; Older people ; Raw Scores ; Schemata (Cognition)</subject><ispartof>Journal of applied research in intellectual disabilities, 2020-09, Vol.33 (5), p.1113-1118</ispartof><rights>2020 The Authors. published by John Wiley & Sons Ltd.</rights><rights>2020 The Authors. Journal of Applied Research in Intellectual Disabilities published by John Wiley & Sons Ltd.</rights><rights>2020. This article is published under http://creativecommons.org/licenses/by-nc/4.0/ (the “License”). Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c4103-6cd18395ef2a71a58cc091ef4b058fc994d1e57f5034ff00522a3fdda9f48173</citedby><cites>FETCH-LOGICAL-c4103-6cd18395ef2a71a58cc091ef4b058fc994d1e57f5034ff00522a3fdda9f48173</cites><orcidid>0000-0002-3041-5253</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://onlinelibrary.wiley.com/doi/pdf/10.1111%2Fjar.12733$$EPDF$$P50$$Gwiley$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1111%2Fjar.12733$$EHTML$$P50$$Gwiley$$Hfree_for_read</linktohtml><link.rule.ids>314,776,780,1411,27901,27902,45550,45551</link.rule.ids><backlink>$$Uhttp://eric.ed.gov/ERICWebPortal/detail?accno=EJ1262944$$DView record in ERIC$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/32281257$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Sauna‐aho, Oili</creatorcontrib><creatorcontrib>Bjelogrlic‐Laakso, Nina</creatorcontrib><creatorcontrib>Rautava, Päivi</creatorcontrib><creatorcontrib>Arvio, Maria</creatorcontrib><title>Ageing and cognition in men with fragile X syndrome</title><title>Journal of applied research in intellectual disabilities</title><addtitle>J Appl Res Intellect Disabil</addtitle><description>Background
Fragile X syndrome (FXS) is the most common inherited cause of intellectual disability. The aim of our longitudinal study was to describe ageing‐related cognitive changes in men with FXS.
Method
A neuropsychologist determined the raw scores (RSs) of 19 men with FXS twice with the Leiter International Performance Scale at an average interval of 22 years. The ages of the participants at baseline ranged from 16 to 50 (mean 27) years.
Results
At follow‐up, the RSs improved in two men, remained the same in two men and declined in 15 men. Overall, the RS of the study group deteriorated by an average 4 points in RSs (p < .001).
Conclusion
Cognitive ageing in men with FXS started earlier than that in men in the general population; in many cases, cognitive ageing in men with FXS began before middle age, usually without any medical or other underlying cause.</description><subject>Adolescents</subject><subject>Adults</subject><subject>ageing</subject><subject>Aging</subject><subject>Aging (Individuals)</subject><subject>Cognitive Ability</subject><subject>cognitive development</subject><subject>fragile X syndrome</subject><subject>Genetic disorders</subject><subject>Intellectual disabilities</subject><subject>Intellectual Disability</subject><subject>Longitudinal Studies</subject><subject>Males</subject><subject>Mens health</subject><subject>Neuropsychology</subject><subject>Older people</subject><subject>Raw Scores</subject><subject>Schemata (Cognition)</subject><issn>1360-2322</issn><issn>1468-3148</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2020</creationdate><recordtype>article</recordtype><sourceid>24P</sourceid><recordid>eNp1kE1LAzEQhoMotlYP_gBlwYsets0k2d3ssZT6UQqC9OAtpNmkpnSzNWkp_fembvUgmMuEzMO8mQeha8B9iGewlL4PpKD0BHWB5TylwPhpvNMcp4QS0kEXISwxxhyy4hx14hMHkhVdRIcLbd0ika5KVLNwdmMbl1iX1NolO7v5SIyXC7vSyXsS9q7yTa0v0ZmRq6CvjrWHZo_j2eg5nb4-vYyG01QxwDTNVQWclpk2RBYgM64ULkEbNscZN6osWQU6K0yGKTMG44wQSU1VydIwDgXtoft27No3n1sdNqK2QenVSjrdbIMglJcEgLM8ond_0GWz9S5-ThBGcVnExVmkHlpK-SYEr41Ye1tLvxeAxUGkiCLFt8jI3h4nbue1rn7JH3MRuGkB7a36bY8nQHJSskPYoO3vorz9_0liMnxrI78ArZ-DMA</recordid><startdate>202009</startdate><enddate>202009</enddate><creator>Sauna‐aho, Oili</creator><creator>Bjelogrlic‐Laakso, Nina</creator><creator>Rautava, Päivi</creator><creator>Arvio, Maria</creator><general>Wiley</general><general>Wiley Subscription Services, Inc</general><scope>24P</scope><scope>7SW</scope><scope>BJH</scope><scope>BNH</scope><scope>BNI</scope><scope>BNJ</scope><scope>BNO</scope><scope>ERI</scope><scope>PET</scope><scope>REK</scope><scope>WWN</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>ASE</scope><scope>FPQ</scope><scope>K6X</scope><scope>K9.</scope><scope>NAPCQ</scope><scope>7X8</scope><orcidid>https://orcid.org/0000-0002-3041-5253</orcidid></search><sort><creationdate>202009</creationdate><title>Ageing and cognition in men with fragile X syndrome</title><author>Sauna‐aho, Oili ; Bjelogrlic‐Laakso, Nina ; Rautava, Päivi ; Arvio, Maria</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c4103-6cd18395ef2a71a58cc091ef4b058fc994d1e57f5034ff00522a3fdda9f48173</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2020</creationdate><topic>Adolescents</topic><topic>Adults</topic><topic>ageing</topic><topic>Aging</topic><topic>Aging (Individuals)</topic><topic>Cognitive Ability</topic><topic>cognitive development</topic><topic>fragile X syndrome</topic><topic>Genetic disorders</topic><topic>Intellectual disabilities</topic><topic>Intellectual Disability</topic><topic>Longitudinal Studies</topic><topic>Males</topic><topic>Mens health</topic><topic>Neuropsychology</topic><topic>Older people</topic><topic>Raw Scores</topic><topic>Schemata (Cognition)</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Sauna‐aho, Oili</creatorcontrib><creatorcontrib>Bjelogrlic‐Laakso, Nina</creatorcontrib><creatorcontrib>Rautava, Päivi</creatorcontrib><creatorcontrib>Arvio, Maria</creatorcontrib><collection>Wiley Online Library Open Access</collection><collection>ERIC</collection><collection>ERIC (Ovid)</collection><collection>ERIC</collection><collection>ERIC</collection><collection>ERIC (Legacy Platform)</collection><collection>ERIC( SilverPlatter )</collection><collection>ERIC</collection><collection>ERIC PlusText (Legacy Platform)</collection><collection>Education Resources Information Center (ERIC)</collection><collection>ERIC</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>British Nursing Index</collection><collection>British Nursing Index (BNI) (1985 to Present)</collection><collection>British Nursing Index</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Nursing & Allied Health Premium</collection><collection>MEDLINE - Academic</collection><jtitle>Journal of applied research in intellectual disabilities</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Sauna‐aho, Oili</au><au>Bjelogrlic‐Laakso, Nina</au><au>Rautava, Päivi</au><au>Arvio, Maria</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><ericid>EJ1262944</ericid><atitle>Ageing and cognition in men with fragile X syndrome</atitle><jtitle>Journal of applied research in intellectual disabilities</jtitle><addtitle>J Appl Res Intellect Disabil</addtitle><date>2020-09</date><risdate>2020</risdate><volume>33</volume><issue>5</issue><spage>1113</spage><epage>1118</epage><pages>1113-1118</pages><issn>1360-2322</issn><eissn>1468-3148</eissn><abstract>Background
Fragile X syndrome (FXS) is the most common inherited cause of intellectual disability. The aim of our longitudinal study was to describe ageing‐related cognitive changes in men with FXS.
Method
A neuropsychologist determined the raw scores (RSs) of 19 men with FXS twice with the Leiter International Performance Scale at an average interval of 22 years. The ages of the participants at baseline ranged from 16 to 50 (mean 27) years.
Results
At follow‐up, the RSs improved in two men, remained the same in two men and declined in 15 men. Overall, the RS of the study group deteriorated by an average 4 points in RSs (p < .001).
Conclusion
Cognitive ageing in men with FXS started earlier than that in men in the general population; in many cases, cognitive ageing in men with FXS began before middle age, usually without any medical or other underlying cause.</abstract><cop>England</cop><pub>Wiley</pub><pmid>32281257</pmid><doi>10.1111/jar.12733</doi><tpages>6</tpages><orcidid>https://orcid.org/0000-0002-3041-5253</orcidid><oa>free_for_read</oa></addata></record> |
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source | Education Source; Wiley Online Library Journals Frontfile Complete |
subjects | Adolescents Adults ageing Aging Aging (Individuals) Cognitive Ability cognitive development fragile X syndrome Genetic disorders Intellectual disabilities Intellectual Disability Longitudinal Studies Males Mens health Neuropsychology Older people Raw Scores Schemata (Cognition) |
title | Ageing and cognition in men with fragile X syndrome |
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