Cargando…

Delineation of early attentional control difficulties in fragile X syndrome: Focus on neurocomputational changes

Fragile X syndrome (FXS) is due to the silencing of a single X-linked gene and it is associated with striking attentional difficulties. As FXS is well characterised at the cellular level, the condition provides a unique opportunity to investigate how a genetic dysfunction can impact on the developme...

Descripción completa

Detalles Bibliográficos
Autores principales: Scerif, Gaia, Cornish, Kim, Wilding, John, Driver, Jon, Karmiloff-Smith, Annette
Formato: Texto
Lenguaje:English
Publicado: Pergamon Press 2007
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2613507/
https://www.ncbi.nlm.nih.gov/pubmed/17254617
http://dx.doi.org/10.1016/j.neuropsychologia.2006.12.005
_version_ 1782163187414597632
author Scerif, Gaia
Cornish, Kim
Wilding, John
Driver, Jon
Karmiloff-Smith, Annette
author_facet Scerif, Gaia
Cornish, Kim
Wilding, John
Driver, Jon
Karmiloff-Smith, Annette
author_sort Scerif, Gaia
collection PubMed
description Fragile X syndrome (FXS) is due to the silencing of a single X-linked gene and it is associated with striking attentional difficulties. As FXS is well characterised at the cellular level, the condition provides a unique opportunity to investigate how a genetic dysfunction can impact on the development of neurocomputational properties relevant to attention. Thirteen young boys with FXS and 13 mental-age-matched typically developing controls performed a touch-screen-based search task that manipulated the similarity between targets and distractors and their heterogeneity in size. Search speed, path and errors were recorded as multiple measures of performance. Children did not differ in overall search speed or path when searching amongst distractors, but striking error patterns distinguished children with FXS from controls. Firstly, although clear markers of previously found targets remained on screen, children with FXS perseverated on touching previous hits more than typically developing controls, consistent with the well-documented inhibitory deficits in adults with the disorder. Secondly, they could accurately discriminate single target-distractor pairs, but, when searching a complex display, they touched distractors more often than control children when distractors were similar to targets and especially so when these were infrequent, highlighting difficulties in judging relative size and allocate attentional weight independently of stimulus frequency. Thirdly, their performance was also characterised by inaccuracies in pointing, suggesting additional motor control deficits. Taken together, the findings suggest that fragile X syndrome affects the early development of multiple processes contributing to efficient attentional selection, as would be predicted from an understanding of the neurocomputational changes associated with the disorder.
format Text
id pubmed-2613507
institution National Center for Biotechnology Information
language English
publishDate 2007
publisher Pergamon Press
record_format MEDLINE/PubMed
spelling pubmed-26135072009-01-05 Delineation of early attentional control difficulties in fragile X syndrome: Focus on neurocomputational changes Scerif, Gaia Cornish, Kim Wilding, John Driver, Jon Karmiloff-Smith, Annette Neuropsychologia Article Fragile X syndrome (FXS) is due to the silencing of a single X-linked gene and it is associated with striking attentional difficulties. As FXS is well characterised at the cellular level, the condition provides a unique opportunity to investigate how a genetic dysfunction can impact on the development of neurocomputational properties relevant to attention. Thirteen young boys with FXS and 13 mental-age-matched typically developing controls performed a touch-screen-based search task that manipulated the similarity between targets and distractors and their heterogeneity in size. Search speed, path and errors were recorded as multiple measures of performance. Children did not differ in overall search speed or path when searching amongst distractors, but striking error patterns distinguished children with FXS from controls. Firstly, although clear markers of previously found targets remained on screen, children with FXS perseverated on touching previous hits more than typically developing controls, consistent with the well-documented inhibitory deficits in adults with the disorder. Secondly, they could accurately discriminate single target-distractor pairs, but, when searching a complex display, they touched distractors more often than control children when distractors were similar to targets and especially so when these were infrequent, highlighting difficulties in judging relative size and allocate attentional weight independently of stimulus frequency. Thirdly, their performance was also characterised by inaccuracies in pointing, suggesting additional motor control deficits. Taken together, the findings suggest that fragile X syndrome affects the early development of multiple processes contributing to efficient attentional selection, as would be predicted from an understanding of the neurocomputational changes associated with the disorder. Pergamon Press 2007 /pmc/articles/PMC2613507/ /pubmed/17254617 http://dx.doi.org/10.1016/j.neuropsychologia.2006.12.005 Text en © 2007 Elsevier Ltd. https://creativecommons.org/licenses/by/3.0/ Open Access under CC BY 3.0 (https://creativecommons.org/licenses/by/3.0/) license
spellingShingle Article
Scerif, Gaia
Cornish, Kim
Wilding, John
Driver, Jon
Karmiloff-Smith, Annette
Delineation of early attentional control difficulties in fragile X syndrome: Focus on neurocomputational changes
title Delineation of early attentional control difficulties in fragile X syndrome: Focus on neurocomputational changes
title_full Delineation of early attentional control difficulties in fragile X syndrome: Focus on neurocomputational changes
title_fullStr Delineation of early attentional control difficulties in fragile X syndrome: Focus on neurocomputational changes
title_full_unstemmed Delineation of early attentional control difficulties in fragile X syndrome: Focus on neurocomputational changes
title_short Delineation of early attentional control difficulties in fragile X syndrome: Focus on neurocomputational changes
title_sort delineation of early attentional control difficulties in fragile x syndrome: focus on neurocomputational changes
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC2613507/
https://www.ncbi.nlm.nih.gov/pubmed/17254617
http://dx.doi.org/10.1016/j.neuropsychologia.2006.12.005
work_keys_str_mv AT scerifgaia delineationofearlyattentionalcontroldifficultiesinfragilexsyndromefocusonneurocomputationalchanges
AT cornishkim delineationofearlyattentionalcontroldifficultiesinfragilexsyndromefocusonneurocomputationalchanges
AT wildingjohn delineationofearlyattentionalcontroldifficultiesinfragilexsyndromefocusonneurocomputationalchanges
AT driverjon delineationofearlyattentionalcontroldifficultiesinfragilexsyndromefocusonneurocomputationalchanges
AT karmiloffsmithannette delineationofearlyattentionalcontroldifficultiesinfragilexsyndromefocusonneurocomputationalchanges