Mapping developmental trajectories of attention and working memory in fragile X syndrome: developmental freeze or developmental change?

Kim Marie Cornish, Victoria Cole, Elena Longhi, Annette Karmiloff-Smith, Gaia Scerif

Research output: Contribution to journalArticleResearchpeer-review

29 Citations (Scopus)

Abstract

Fragile X syndrome (FXS) has a characteristic cognitive signature that by late childhood includes core weaknesses in attention and working memory (WM), but their earlier developmental trajectories remain uncharted. Using a combined cross-sectional and prospective longitudinal design, we tested whether early profiles of attention and WM impairment in FXS indicate developmental freeze or developmental change. In Study 1, 26 young boys with FXS and 55 typically developing (TD) boys completed two experimental paradigms designed to assess cognitive aspects of attention and WM, in addition to behavioral indices of inattention and hyperactivity. Study 2 mapped longitudinal changes in 21 children with FXS and 21 TD children. In Study 1, significant weaknesses emerged for boys with FXS, with no substantial improvement over chronological age. Mapping performance against mental age level revealed delay, but it also yielded a similar attention and WM profile to TD boys. In Study 2, longitudinal improvements for boys with FXS paralleled those in TD children. In conclusion, cognitive attention and WM, although delayed in FXS, reveal developmental change, rather than arrest. Our findings underscore the need for going beyond cross-sectional group comparisons and gross behavioral indices to map cognitive changes longitudinally in developmental disorders.
Original languageEnglish
Pages (from-to)365 - 376
Number of pages12
JournalDevelopment and Psychopathology
Volume25
Issue number2
DOIs
Publication statusPublished - 2013

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