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Improving social gaze behavior in fragile X syndrome using a behavioral skills training approach: a proof of concept study
Journal of Neurodevelopmental Disorders ( IF 4.9 ) Pub Date : 2018-08-28 , DOI: 10.1186/s11689-018-9243-z
Caitlin E. Gannon , Tobias C. Britton , Ellen H. Wilkinson , Scott S. Hall

Individuals diagnosed with fragile X syndrome (FXS), the most common known inherited form of intellectual disability, commonly exhibit significant impairments in social gaze behavior during interactions with others. Although this behavior can restrict social development and limit educational opportunities, behavioral interventions designed to improve social gaze behavior have not been developed for this population. In this proof of concept (PoC) study, we examined whether administering a behavioral skills training package—discrete trial instruction (DTI) plus relaxation training—could increase social gaze duration in males with FXS. As part of a larger clinical trial, 20 boys with FXS, aged 8 to 18 years, were randomized to receive DTI plus relaxation training administered at one of two prescribed doses over a 2-day period at our research center. Potential improvements in social gaze behavior were evaluated by direct observations conducted across trials during the training, and generalization effects were examined by administering a social challenge before and after the treatment. During the social challenge, social gaze behavior was recorded using an eye tracker and physiological arousal levels were simultaneously recorded by monitoring the child’s heart rate. Levels of social gaze behavior increased significantly across blocks of training trials for six (60%) boys who received the high-dose behavioral treatment and for three (30%) boys who received the low-dose behavioral treatment. Boys who received the high-dose treatment also showed greater improvements in social gaze behavior during the social challenge compared to boys who received the low-dose treatment. There was no effect of the treatment on physiological arousal levels recorded on the heart rate monitor at either dose. These results suggest that appropriate social gaze behavior can be successfully taught to boys with FXS using a standardized behavioral skills training approach. Future studies will need to evaluate whether younger children with FXS might benefit from this treatment, and/or whether more naturalistic forms of behavioral skills training might be beneficial, before social gaze avoidance becomes established in the child’s repertoire. ClinicalTrials.gov, NCT02616796 . Registered 30 November 2015.

中文翻译:

使用行为技能训练方法改善脆性X综合征的社交注视行为:概念验证

被诊断为脆性X综合征(FXS)(最常见的已知智力障碍遗传形式)的个体在与他人互动时通常会表现出社交注视行为的重大损害。尽管这种行为会限制社会发展并限制教育机会,但尚未针对该人群开发旨在改善社交注视行为的行为干预措施。在这项概念验证(PoC)研究中,我们研究了管理行为技能培训包(离散试验指导(DTI)和放松训练)是否会增加FXS男性的社交注视持续时间。作为一项较大的临床试验的一部分,我们的研究中心在2天的时间内将20名年龄在8至18岁的FXS男孩随机接受两种规定剂量之一的DTI加放松训练。通过在培训期间跨试验进行的直接观察来评估社交注视行为的潜在改善,并通过在治疗之前和之后进行社交挑战来检查泛化效果。在社交挑战中,使用眼动仪记录社交注视行为,并通过监视孩子的心率同时记录生理唤醒水平。在六个训练试验中,接受高剂量行为治疗的男孩(60%)和接受低剂量行为治疗的三个男孩(30%),在整个训练试验中,社交注视行为的水平显着提高。与接受低剂量治疗的男孩相比,接受高剂量治疗的男孩在社交挑战中的社交注视行为也表现出更大的改善。在任何剂量下,该治疗对心率监测器上记录的生理唤醒水平均无影响。这些结果表明,可以使用标准化的行为技能培训方法,向患有FXS的男孩成功地教授适当的社交凝视行为。未来的研究将需要评估,在避开社交注视之前,更年轻的FXS儿童可能会从这种治疗中受益,和/或更自然的形式的行为技能培训是否会受益。ClinicalTrials.gov,NCT02616796。2015年11月30日注册。这些结果表明,可以使用标准化的行为技能培训方法,向患有FXS的男孩成功地教授适当的社交凝视行为。未来的研究将需要评估,在避开社交注视之前,更年轻的FXS儿童可能会从这种治疗中受益,和/或更自然的形式的行为技能培训是否会受益。ClinicalTrials.gov,NCT02616796。2015年11月30日注册。这些结果表明,可以使用标准化的行为技能培训方法,向患有FXS的男孩成功地教授适当的社交凝视行为。未来的研究将需要评估,在避开社交注视之前,在这种情况下,年龄较小的FXS儿童是否可能会从这种治疗中受益,和/或更自然的行为技能培训形式是否会有所裨益。ClinicalTrials.gov,NCT02616796。2015年11月30日注册。避免在孩子的曲目中建立社交注视。ClinicalTrials.gov,NCT02616796。2015年11月30日注册。避免在孩子的曲目中建立社交注视。ClinicalTrials.gov,NCT02616796。2015年11月30日注册。
更新日期:2018-08-28
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