Early gesture use in fragile X syndrome

Background Emerging evidence suggests that children with fragile X syndrome (FXS) exhibit abnormal gesture use early in development, although few studies have investigated the emergence of gesture use in this population or the impact of autism spectrum disorder (ASD) features on these behaviours. Th...

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Veröffentlicht in:Journal of intellectual disability research 2018-07, Vol.62 (7), p.625-636
Hauptverfasser: Rague, L., Caravella, K., Tonnsen, B., Klusek, J., Roberts, J.
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container_end_page 636
container_issue 7
container_start_page 625
container_title Journal of intellectual disability research
container_volume 62
creator Rague, L.
Caravella, K.
Tonnsen, B.
Klusek, J.
Roberts, J.
description Background Emerging evidence suggests that children with fragile X syndrome (FXS) exhibit abnormal gesture use early in development, although few studies have investigated the emergence of gesture use in this population or the impact of autism spectrum disorder (ASD) features on these behaviours. The present study examined the longitudinal development of gesture use in infants with FXS relative to low‐risk controls and infant siblings of children with ASD (high‐risk siblings), with the goal of establishing potentially unique patterns of gesture development in infants with FXS and understanding the relative impact of ASD symptom severity on these patterns. Method Participants included 86 male infants (39 FXS, 27 high‐risk siblings and 20 low‐risk infants) assessed at 9, 12 and 24 months of age. Multilevel modelling was used to assess differences in number of gestures used and rates of gesture use across groups, as well as the relative impact of ASD symptom severity and nonverbal skills on these patterns. Results Infants with FXS used fewer gestures than high‐risk siblings and low‐risk infants, with this difference being primarily accounted for by the effect of low nonverbal abilities in the FXS group. Furthermore, although higher ASD symptom severity was associated with the use of fewer gestures in both the FXS and high‐risk sibling groups, a significant amount of variance was shared between ASD symptom severity and nonverbal skills in FXS, but not in high‐risk siblings. Conclusions This study presents the first longitudinal analysis of early gesture development in FXS by using a multigroup design, clarifying the relative roles of cognitive deficits and ASD symptom severity in the development of gesture use in FXS. These findings offer novel evidence that early gesture use in FXS may reflect broader features of the FXS phenotype rather than predicting later social‐communicative deficits characteristic of comorbid ASD.
doi_str_mv 10.1111/jir.12498
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The present study examined the longitudinal development of gesture use in infants with FXS relative to low‐risk controls and infant siblings of children with ASD (high‐risk siblings), with the goal of establishing potentially unique patterns of gesture development in infants with FXS and understanding the relative impact of ASD symptom severity on these patterns. Method Participants included 86 male infants (39 FXS, 27 high‐risk siblings and 20 low‐risk infants) assessed at 9, 12 and 24 months of age. Multilevel modelling was used to assess differences in number of gestures used and rates of gesture use across groups, as well as the relative impact of ASD symptom severity and nonverbal skills on these patterns. Results Infants with FXS used fewer gestures than high‐risk siblings and low‐risk infants, with this difference being primarily accounted for by the effect of low nonverbal abilities in the FXS group. Furthermore, although higher ASD symptom severity was associated with the use of fewer gestures in both the FXS and high‐risk sibling groups, a significant amount of variance was shared between ASD symptom severity and nonverbal skills in FXS, but not in high‐risk siblings. Conclusions This study presents the first longitudinal analysis of early gesture development in FXS by using a multigroup design, clarifying the relative roles of cognitive deficits and ASD symptom severity in the development of gesture use in FXS. These findings offer novel evidence that early gesture use in FXS may reflect broader features of the FXS phenotype rather than predicting later social‐communicative deficits characteristic of comorbid ASD.</description><identifier>ISSN: 0964-2633</identifier><identifier>EISSN: 1365-2788</identifier><identifier>DOI: 10.1111/jir.12498</identifier><identifier>PMID: 29781139</identifier><language>eng</language><publisher>England: Wiley Subscription Services, Inc</publisher><subject>Autism ; Autism Spectrum Disorder - complications ; Autism Spectrum Disorder - physiopathology ; Autism Spectrum Disorder - psychology ; Autism Spectrum Disorders ; Autistic children ; behavioural phenotypes ; Child Behavior - psychology ; Child, Preschool ; Cognitive impairment ; communication ; Comorbidity ; Follow-Up Studies ; fragile X ; Fragile X syndrome ; Fragile X Syndrome - complications ; Fragile X Syndrome - physiopathology ; Fragile X Syndrome - psychology ; gesture ; Gestures ; Humans ; Infant ; Infants ; Longitudinal Studies ; Male ; Nonverbal Ability ; Risk assessment ; Severity ; Severity of Illness Index ; Siblings ; Siblings - psychology ; Young Children</subject><ispartof>Journal of intellectual disability research, 2018-07, Vol.62 (7), p.625-636</ispartof><rights>2018 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and John Wiley &amp; Sons Ltd</rights><rights>2018 MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and John Wiley &amp; Sons Ltd.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c4438-9ff1d7d63e96f7227c99b3df1d5e8d0dfa2ee1dca25072cf29ac625625e3672a3</citedby><cites>FETCH-LOGICAL-c4438-9ff1d7d63e96f7227c99b3df1d5e8d0dfa2ee1dca25072cf29ac625625e3672a3</cites><orcidid>0000-0002-8372-5837 ; 0000-0001-7966-1837</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%2Fjir.12498$$EPDF$$P50$$Gwiley$$H</linktopdf><linktohtml>$$Uhttps://onlinelibrary.wiley.com/doi/full/10.1111%2Fjir.12498$$EHTML$$P50$$Gwiley$$H</linktohtml><link.rule.ids>230,314,780,784,885,1417,27924,27925,30999,45574,45575</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/29781139$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Rague, L.</creatorcontrib><creatorcontrib>Caravella, K.</creatorcontrib><creatorcontrib>Tonnsen, B.</creatorcontrib><creatorcontrib>Klusek, J.</creatorcontrib><creatorcontrib>Roberts, J.</creatorcontrib><title>Early gesture use in fragile X syndrome</title><title>Journal of intellectual disability research</title><addtitle>J Intellect Disabil Res</addtitle><description>Background Emerging evidence suggests that children with fragile X syndrome (FXS) exhibit abnormal gesture use early in development, although few studies have investigated the emergence of gesture use in this population or the impact of autism spectrum disorder (ASD) features on these behaviours. The present study examined the longitudinal development of gesture use in infants with FXS relative to low‐risk controls and infant siblings of children with ASD (high‐risk siblings), with the goal of establishing potentially unique patterns of gesture development in infants with FXS and understanding the relative impact of ASD symptom severity on these patterns. Method Participants included 86 male infants (39 FXS, 27 high‐risk siblings and 20 low‐risk infants) assessed at 9, 12 and 24 months of age. Multilevel modelling was used to assess differences in number of gestures used and rates of gesture use across groups, as well as the relative impact of ASD symptom severity and nonverbal skills on these patterns. Results Infants with FXS used fewer gestures than high‐risk siblings and low‐risk infants, with this difference being primarily accounted for by the effect of low nonverbal abilities in the FXS group. Furthermore, although higher ASD symptom severity was associated with the use of fewer gestures in both the FXS and high‐risk sibling groups, a significant amount of variance was shared between ASD symptom severity and nonverbal skills in FXS, but not in high‐risk siblings. Conclusions This study presents the first longitudinal analysis of early gesture development in FXS by using a multigroup design, clarifying the relative roles of cognitive deficits and ASD symptom severity in the development of gesture use in FXS. These findings offer novel evidence that early gesture use in FXS may reflect broader features of the FXS phenotype rather than predicting later social‐communicative deficits characteristic of comorbid ASD.</description><subject>Autism</subject><subject>Autism Spectrum Disorder - complications</subject><subject>Autism Spectrum Disorder - physiopathology</subject><subject>Autism Spectrum Disorder - psychology</subject><subject>Autism Spectrum Disorders</subject><subject>Autistic children</subject><subject>behavioural phenotypes</subject><subject>Child Behavior - psychology</subject><subject>Child, Preschool</subject><subject>Cognitive impairment</subject><subject>communication</subject><subject>Comorbidity</subject><subject>Follow-Up Studies</subject><subject>fragile X</subject><subject>Fragile X syndrome</subject><subject>Fragile X Syndrome - complications</subject><subject>Fragile X Syndrome - physiopathology</subject><subject>Fragile X Syndrome - psychology</subject><subject>gesture</subject><subject>Gestures</subject><subject>Humans</subject><subject>Infant</subject><subject>Infants</subject><subject>Longitudinal Studies</subject><subject>Male</subject><subject>Nonverbal Ability</subject><subject>Risk assessment</subject><subject>Severity</subject><subject>Severity of Illness Index</subject><subject>Siblings</subject><subject>Siblings - psychology</subject><subject>Young Children</subject><issn>0964-2633</issn><issn>1365-2788</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2018</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><sourceid>7QJ</sourceid><recordid>eNp1kF1LwzAUhoMobk4v_ANS8EK96JacNGlzI8iYOhEEUfAuZGk6O_oxk1Xpvzezc6hgOBA45-HhnBehY4KHxL_RIrdDApFIdlCfUM5CiJNkF_Wx4FEInNIeOnBugTHmJOL7qAciTgihoo_OJsoWbTA3btVYEzTOBHkVZFbN88IEL4Frq9TWpTlEe5kqnDna_AP0fD15Gt-G9w830_HVfaijiCahyDKSximnRvAsBoi1EDOa-iYzSYrTTIExJNUKGI5BZyCU5sB8GcpjUHSALjvvspmVJtWmWllVyKXNS2VbWatc_p5U-auc1--SCwaECC843whs_db4s2SZO22KQlWmbpwEHAFEGMfMo6d_0EXd2Mqf5ymGWQQJXlMXHaVt7Zw12XYZguU6funjl1_xe_bk5_Zb8jtvD4w64MPH2_5vknfTx075CXiVjgc</recordid><startdate>201807</startdate><enddate>201807</enddate><creator>Rague, L.</creator><creator>Caravella, K.</creator><creator>Tonnsen, B.</creator><creator>Klusek, J.</creator><creator>Roberts, J.</creator><general>Wiley Subscription Services, Inc</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7QJ</scope><scope>7X8</scope><scope>5PM</scope><orcidid>https://orcid.org/0000-0002-8372-5837</orcidid><orcidid>https://orcid.org/0000-0001-7966-1837</orcidid></search><sort><creationdate>201807</creationdate><title>Early gesture use in fragile X syndrome</title><author>Rague, L. ; Caravella, K. ; Tonnsen, B. ; Klusek, J. ; Roberts, J.</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c4438-9ff1d7d63e96f7227c99b3df1d5e8d0dfa2ee1dca25072cf29ac625625e3672a3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2018</creationdate><topic>Autism</topic><topic>Autism Spectrum Disorder - complications</topic><topic>Autism Spectrum Disorder - physiopathology</topic><topic>Autism Spectrum Disorder - psychology</topic><topic>Autism Spectrum Disorders</topic><topic>Autistic children</topic><topic>behavioural phenotypes</topic><topic>Child Behavior - psychology</topic><topic>Child, Preschool</topic><topic>Cognitive impairment</topic><topic>communication</topic><topic>Comorbidity</topic><topic>Follow-Up Studies</topic><topic>fragile X</topic><topic>Fragile X syndrome</topic><topic>Fragile X Syndrome - complications</topic><topic>Fragile X Syndrome - physiopathology</topic><topic>Fragile X Syndrome - psychology</topic><topic>gesture</topic><topic>Gestures</topic><topic>Humans</topic><topic>Infant</topic><topic>Infants</topic><topic>Longitudinal Studies</topic><topic>Male</topic><topic>Nonverbal Ability</topic><topic>Risk assessment</topic><topic>Severity</topic><topic>Severity of Illness Index</topic><topic>Siblings</topic><topic>Siblings - psychology</topic><topic>Young Children</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Rague, L.</creatorcontrib><creatorcontrib>Caravella, K.</creatorcontrib><creatorcontrib>Tonnsen, B.</creatorcontrib><creatorcontrib>Klusek, J.</creatorcontrib><creatorcontrib>Roberts, J.</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>Applied Social Sciences Index &amp; Abstracts (ASSIA)</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Journal of intellectual disability research</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Rague, L.</au><au>Caravella, K.</au><au>Tonnsen, B.</au><au>Klusek, J.</au><au>Roberts, J.</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Early gesture use in fragile X syndrome</atitle><jtitle>Journal of intellectual disability research</jtitle><addtitle>J Intellect Disabil Res</addtitle><date>2018-07</date><risdate>2018</risdate><volume>62</volume><issue>7</issue><spage>625</spage><epage>636</epage><pages>625-636</pages><issn>0964-2633</issn><eissn>1365-2788</eissn><abstract>Background Emerging evidence suggests that children with fragile X syndrome (FXS) exhibit abnormal gesture use early in development, although few studies have investigated the emergence of gesture use in this population or the impact of autism spectrum disorder (ASD) features on these behaviours. The present study examined the longitudinal development of gesture use in infants with FXS relative to low‐risk controls and infant siblings of children with ASD (high‐risk siblings), with the goal of establishing potentially unique patterns of gesture development in infants with FXS and understanding the relative impact of ASD symptom severity on these patterns. Method Participants included 86 male infants (39 FXS, 27 high‐risk siblings and 20 low‐risk infants) assessed at 9, 12 and 24 months of age. Multilevel modelling was used to assess differences in number of gestures used and rates of gesture use across groups, as well as the relative impact of ASD symptom severity and nonverbal skills on these patterns. Results Infants with FXS used fewer gestures than high‐risk siblings and low‐risk infants, with this difference being primarily accounted for by the effect of low nonverbal abilities in the FXS group. Furthermore, although higher ASD symptom severity was associated with the use of fewer gestures in both the FXS and high‐risk sibling groups, a significant amount of variance was shared between ASD symptom severity and nonverbal skills in FXS, but not in high‐risk siblings. Conclusions This study presents the first longitudinal analysis of early gesture development in FXS by using a multigroup design, clarifying the relative roles of cognitive deficits and ASD symptom severity in the development of gesture use in FXS. These findings offer novel evidence that early gesture use in FXS may reflect broader features of the FXS phenotype rather than predicting later social‐communicative deficits characteristic of comorbid ASD.</abstract><cop>England</cop><pub>Wiley Subscription Services, Inc</pub><pmid>29781139</pmid><doi>10.1111/jir.12498</doi><tpages>12</tpages><orcidid>https://orcid.org/0000-0002-8372-5837</orcidid><orcidid>https://orcid.org/0000-0001-7966-1837</orcidid><oa>free_for_read</oa></addata></record>
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subjects Autism
Autism Spectrum Disorder - complications
Autism Spectrum Disorder - physiopathology
Autism Spectrum Disorder - psychology
Autism Spectrum Disorders
Autistic children
behavioural phenotypes
Child Behavior - psychology
Child, Preschool
Cognitive impairment
communication
Comorbidity
Follow-Up Studies
fragile X
Fragile X syndrome
Fragile X Syndrome - complications
Fragile X Syndrome - physiopathology
Fragile X Syndrome - psychology
gesture
Gestures
Humans
Infant
Infants
Longitudinal Studies
Male
Nonverbal Ability
Risk assessment
Severity
Severity of Illness Index
Siblings
Siblings - psychology
Young Children
title Early gesture use in fragile X syndrome
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