Attention, Brain and Cognitive Development Group, Department of Experimental Psychology, University of Oxford, Oxford, UK.
J Child Psychol Psychiatry. 2012 Jun;53(6):641-50. doi: 10.1111/j.1469-7610.2011.02515.x. Epub 2011 Dec 29.
Fragile X syndrome (FXS) is an early diagnosed monogenic disorder, associated with a striking pattern of cognitive/attentional difficulties and a high risk of poor behavioural outcomes. FXS therefore represents an ideal model disorder to study prospectively the impact of early attention deficits on behaviour.
Thirty-seven boys with FXS aged 4-10 years and 74 typically developing (TD) boys took part. Study 1 was designed to assess visual and auditory attention at two time-points, 1 year apart. Study 2 investigated attention to multimodal information. Both tested attention markers as longitudinal predictors of risk for poor behaviour in FXS.
Children with FXS attended less well than mental-age matched TD boys and experienced greater difficulties with auditory compared to visual stimuli. In addition, unlike TD children, they did not benefit from multimodal information. Attention markers were significant predictors of later behavioural difficulties in boys with FXS.
Findings demonstrate, for the first time, greater difficulties with auditory attention and atypical processing of multimodal information, in addition to pervasive global attentional difficulties in boys with FXS. Attention predicted outcomes longitudinally, underscoring the need to dissect what drives differing developmental trajectories for individual children within a seemingly homogeneous group.
脆性 X 综合征（FXS）是一种早期诊断的单基因疾病，与明显的认知/注意力困难模式和不良行为结果的高风险相关。因此，FXS 代表了一个理想的模型障碍，可用于前瞻性研究早期注意力缺陷对行为的影响。
37 名 4-10 岁的 FXS 男孩和 74 名典型发育（TD）男孩参加了这项研究。研究 1 旨在评估视觉和听觉注意力，两次测试时间相隔 1 年。研究 2 研究了对多模态信息的注意力。两者均将注意力标志物作为 FXS 不良行为风险的纵向预测指标进行测试。
与智力年龄匹配的 TD 男孩相比，FXS 患儿的注意力较差，与视觉刺激相比，他们对听觉刺激的反应更差。此外，与 TD 儿童不同，他们没有从多模态信息中受益。注意力标志物是 FXS 男孩日后行为困难的重要预测指标。
这些发现首次证明，与普遍的全局注意力困难相比，FXS 男孩还存在听觉注意力困难和多模态信息处理异常。注意力从纵向预测了结果，强调需要剖析是什么原因导致在看似同质的群体中，每个孩子的发展轨迹不同。
