A 'learning platform' approach to outcome measurement in fragile X syndrome: a preliminary psychometric study
A 'learning platform' approach to outcome measurement in fragile X syndrome: a preliminary psychometric study
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DOI：
10.1111/j.1365-2788.2012.01560.x
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发表时间：
2012-10-01
影响因子：
3.6
通讯作者：
Reiss, A. L.
中科院分区：
文献类型：
--
作者：
Hall, S. S.;Hammond, J. L.;Reiss, A. L.
背景缓解脆性X综合征(FXS)患者认知和行为症状的药物临床试验目前正在进行中。然而，几乎没有可靠、有效和/或敏感的结果衡量标准可以直接用于FXS患者。临床试验中使用的大多数评估对智障(ID)患者可能不是最理想的，因为它们需要与检验员进行面对面的互动，对管理周期造成负担，并且在测试期间不提供强化和/或反馈。因此，我们研究了一种新的计算机化的FXS结果测量方法的心理测量学特性。方法对13名患有FXS的女孩、12名患有FXS的男孩和15名年龄在10至23岁(心理年龄=4至12岁)的匹配ID控制组进行了简短的计算机化测试，并将其纳入离散试验训练器(C)--一种为患有ID的儿童设计的商业软件程序中。该软件提供了每一次试验的增援、反馈、令牌交付和提示程序(如有必要)的自动应急访问，以便于作出反应。主要测量结果是参与者的学习速度，从参与者的正确回答的累积记录中得出。结果所有参与者都能够完成测试，地板效应似乎是最小的。学习率平均为每分钟五个正确回答，每组每分钟一个到八个正确回答。FXS女孩的重测信度为0.77，FXS男孩的重测信度为0.90，匹配的ID对照组的重测信度为0.90。与韦氏儿童智力量表-III算术分测验的原始分数相比，FXS女孩为0.35分，FXS男孩为0.80分，匹配的ID对照组为0.56分。学习效率对变化也高度敏感，经过超过1.5d的15~20次、15分钟的高强度离散试验训练后，每组的有效值分别为1.21、0.89和1.47。结论这些结果表明，学习平台方法可以为研究人员提供一种可靠、有效和高度敏感的方法来评估治疗效果，不仅对FXS患者，而且对其他ID患者。
Background Clinical trials of medications to alleviate the cognitive and behavioural symptoms of individuals with fragile X syndrome (FXS) are now underway. However, there are few reliable, valid and/or sensitive outcome measures available that can be directly administered to individuals with FXS. The majority of assessments employed in clinical trials may be suboptimal for individuals with intellectual disability (ID) because they require face-to-face interaction with an examiner, taxing administration periods, and do not provide reinforcement and/or feedback during the test. We therefore examined the psychometric properties of a new computerised 'learning platform' approach to outcome measurement in FXS.Method A brief computerised test, incorporated into the Discrete Trial Trainer (c) - a commercially available software program designed for children with ID - was administered to 13 girls with FXS, 12 boys with FXS and 15 matched ID controls aged 10 to 23 years (mental age = 4 to 12 years). The software delivered automated contingent access to reinforcement, feedback, token delivery and prompting procedures (if necessary) on each trial to facilitate responding. The primary outcome measure was the participant's learning rate, derived from the participant's cumulative record of correct responses.Results All participants were able to complete the test and floor effects appeared to be minimal. Learning rates averaged approximately five correct responses per minute, ranging from one to eight correct responses per minute in each group. Test-retest reliability of the learning rates was 0.77 for girls with FXS, 0.90 for boys with FXS and 0.90 for matched ID controls. Concurrent validity with raw scores obtained on the Arithmetic subtest of the Wechsler Intelligence Scale for Children-III was 0.35 for girls with FXS, 0.80 for boys with FXS and 0.56 for matched ID controls. The learning rates were also highly sensitive to change, with effect sizes of 1.21, 0.89 and 1.47 in each group respectively following 15 to 20, 15-min sessions of intensive discrete trial training conducted over 1.5 days.Conclusions These results suggest that a learning platform approach to outcome measurement could provide investigators with a reliable, valid and highly sensitive measure to evaluate treatment efficacy, not only for individuals with FXS but also for individuals with other ID.