Department of Allied Health Sciences, 1028 Bondurant Hall, School of Medicine, University of North Carolina School-Chapel Hill, Chapel Hill, North Carolina, 27599-4120, USA.
Department of Special Education, Box 228, Peabody College, Vanderbilt University, Nashville, Tennessee, 37203, USA.
Res Dev Disabil. 2018 Oct;81:73-88. doi: 10.1016/j.ridd.2018.05.014. Epub 2018 Jun 20.
Executive functions (EF) have been identified as impaired in FXS, but few studies have examined their developmental trajectories.
The primary aim of this longitudinal study was to examine the development of EF in young males with FXS compared to Mental Age (MA)-matched controls.
The sample comprised 56 boys with FXS (ages 7-13 years), and 48 MA-matched typical boys (ages 4-8 years). EF tasks included measures of inhibitory control, working memory, cognitive flexibility/set-shifting, problem solving/planning, and processing speed. Tasks were administered at three time points over five-years.
The MA-Matched Typical boys significantly outperformed the FXS boys on all EF tasks, with the FXS Group showing a pattern of slow, but positive growth on most EF tasks. For working memory tasks, significant interactions were noted between MA and autism symptom severity, and MA and medication status. The probability of task completion increased with higher MA.
These findings contribute to our understanding of the development of EF in this population. They also lay the foundation for use of EF tasks in treatment efforts, particularly with respect to documenting improvements and practice effects, and in understanding associations with targeted developmental outcomes.
执行功能（EF）在 FXS 中已被确定为受损，但很少有研究检查其发展轨迹。
本纵向研究的主要目的是比较 FXS 年轻男性与智力年龄（MA）匹配对照组的 EF 发展。
该样本包括 56 名 FXS 男孩（年龄 7-13 岁）和 48 名 MA 匹配的正常男孩（年龄 4-8 岁）。EF 任务包括抑制控制、工作记忆、认知灵活性/定势转换、问题解决/计划和加工速度的测量。任务在五年内分三个时间点进行。
MA 匹配的正常男孩在所有 EF 任务上的表现均明显优于 FXS 男孩，而 FXS 组在大多数 EF 任务上表现出缓慢但积极的增长模式。对于工作记忆任务，在 MA 和自闭症症状严重程度以及 MA 和药物状态之间注意到显著的相互作用。任务完成的可能性随着 MA 的增加而增加。
这些发现有助于我们理解该人群中 EF 的发展。它们还为 EF 任务在治疗中的应用奠定了基础，特别是在记录改善和练习效果方面，以及在理解与有针对性的发展结果的关联方面。
