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

机译：绘制脆弱X综合征中注意力和工作记忆的发展轨迹：发育冻结还是发育变化？

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摘要

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.
机译：脆性X综合征（FXS）具有特征性的认知特征，到儿童晚期时，其注意力和工作记忆（WM）便成为核心弱点，但其早期发展轨迹仍然未知。使用组合的横截面和前瞻性纵向设计，我们测试了FXS中注意力和WM损伤的早期状况是否表明发育冻结或发育变化。在研究1中，除了注意力不集中和活动过度的行为指标外，有26个FXS年轻男孩和55个典型发育中（TD）男孩完成了两个实验范式，旨在评估注意力和WM的认知方面。研究2绘制了21名FXS儿童和21名TD儿童的纵向变化。在研究1中，患有FXS的男孩出现了明显的弱点，但随着年龄的增长没有明显改善。对照心理年龄水平的表现表现出了延迟，但它也引起了与TD男孩相似的关注和WM状况。在研究2中，具有FXS的男孩的纵向改善与TD儿童的平行。总之，认知注意和WM尽管在FXS中有所延迟，但显示出发展变化，而不是停滞。我们的研究结果强调，需要超越横断面组比较和总体行为指标来纵向描述发育障碍中的认知变化。

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