Scott Haleigh, Harvey Danielle J, Li Yueju, McLennan Yingratana A, Johnston Cindy K, Shickman Ryan, Piven Joseph, Schweitzer Julie B, Hessl David
MIND Institute, University of California Davis Medical Center, Sacramento, CA 95817, USA.
Department of Psychiatry and Behavioral Sciences, University of California Davis School of Medicine, Sacramento, CA 95817, USA.
Brain Sci. 2020 Sep 25;10(10):671. doi: 10.3390/brainsci10100671.
Children with fragile X syndrome (FXS) exhibit deficits in a variety of cognitive processes within the executive function domain. As working memory (WM) is known to support a wide range of cognitive, learning and adaptive functions, WM computer-based training programs have the potential to benefit people with FXS and other forms of intellectual and developmental disability (IDD). However, research on the effectiveness of WM training has been mixed. The current study is a follow-up "deep dive" into the data collected during a randomized controlled trial of Cogmed (Stockholm, Sweden) WM training in children with FXS. Analyses characterized the training data, identified training quality metrics, and identified subgroups of participants with similar training patterns. Child, parent, home environment and training quality metrics were explored in relation to the clinical outcomes during the WM training intervention. Baseline cognitive level and training behavior metrics were linked to gains in WM performance-based assessments and also to reductions in inattention and other behaviors related to executive functioning during the intervention. The results also support a recommendation that future cognitive intervention trials with individuals with IDD such as FXS include additional screening of participants to determine not only baseline feasibility, but also capacity for training progress over a short period prior to inclusion and randomization. This practice may also better identify individuals with IDD who are more likely to benefit from cognitive training in clinical and educational settings.
脆性X综合征（FXS）患儿在执行功能领域的多种认知过程中存在缺陷。由于工作记忆（WM）已知可支持广泛的认知、学习和适应功能，基于计算机的WM训练计划有可能使FXS患者以及其他形式的智力和发育障碍（IDD）患者受益。然而，关于WM训练有效性的研究结果不一。当前的研究是对在一项针对FXS患儿的Cogmed（瑞典斯德哥尔摩）WM训练随机对照试验期间收集的数据进行的后续“深入研究”。分析对训练数据进行了特征描述，确定了训练质量指标，并确定了具有相似训练模式的参与者亚组。在WM训练干预期间，探讨了儿童、家长、家庭环境和训练质量指标与临床结果之间的关系。基线认知水平和训练行为指标与基于WM表现的评估中的增益相关，也与干预期间注意力不集中及其他与执行功能相关行为的减少相关。研究结果还支持一项建议，即未来针对FXS等IDD个体的认知干预试验应包括对参与者进行额外筛查，不仅要确定基线可行性，还要在纳入和随机分组之前的短时间内确定训练进展的能力。这种做法还可能更好地识别出在临床和教育环境中更有可能从认知训练中受益的IDD个体。
