Children with fragile X syndrome (FXS) are at increased risk for exhibiting problem behaviors such as aggression and self-injury. However, many children with FXS have limited access to behavioral treatments that have known efficacy due to the low availability of treatment providers and the wide geographical dispersion of families with FXS across the country. Telehealth may offer a cost-effective and practical solution to overcome these significant barriers. We examined the effect of administering an established behavior analytic intervention called functional communication training (FCT) via telehealth on levels of problem behaviors exhibited by boys with FXS. We also examined treatment acceptability, as well as the effect of the treatment on levels of parenting stress. Boys with FXS, aged 3 to 10 years, who displayed problem behaviors daily, were randomized to receive FCT via telehealth (n = 30) or treatment as usual (n = 27) over 12 weeks. Outcome measures included in-session observations of problem behavior, the Aberrant Behavior Checklist—Community (ABC-C), the Treatment Acceptability Rating Form—Revised (TARF-R), and the Parenting Stress Index, 4th edition (PSI-4). Intention-to-treat analyses indicated that scores on the irritability subscale of the ABC-C, our primary outcome measure, decreased significantly for boys who received FCT via telehealth compared to boys who received treatment as usual (p < .001, Cohen’s d = 0.65). In-session observations conducted for those who received treatment showed that levels of problem behavior decreased by 91% from baseline. Levels of parenting stress related to child behavioral problems were also lower following FCT treatment, and caregivers reported that the intervention was acceptable. These findings support telehealth-enabled FCT as a framework for expanding access to behavioral treatments for problem behaviors in children with FXS. Expanded delivery of behavior analytic treatment via telehealth also has the potential to lower healthcare costs, improve child and family quality of life, and lead to advances in the treatment of problem behavior in the broader population of individuals with neurodevelopmental disorders. ClinicalTrials.gov, NCT03510156 . Registered 27 April 2018

中文翻译：

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针对脆性 X 综合征男孩问题行为的远程医疗行为治疗：一项随机对照试验

患有脆性 X 综合征 (FXS) 的儿童表现出攻击性和自伤等问题行为的风险更高。然而，由于治疗提供者的可用性低以及 FXS 家庭在全国范围内的广泛地理分布，许多 FXS 儿童获得已知疗效的行为治疗的机会有限。远程医疗可能会提供一种经济高效且实用的解决方案来克服这些重大障碍。我们研究了通过远程医疗管理一种名为功能性沟通训练 (FCT) 的既定行为分析干预对患有 FXS 的男孩表现出的问题行为水平的影响。我们还检查了治疗的可接受性，以及治疗对育儿压力水平的影响。患有 FXS 的男孩，年龄在 3 到 10 岁之间，每天都表现出问题行为，随机接受远程医疗（n = 30）或常规治疗（n = 27）超过 12 周的 FCT。结果测量包括对问题行为的会话观察、异常行为检查表 - 社区 (ABC-C)、治疗可接受性评级表 - 修订版 (TARF-R) 和育儿压力指数，第 4 版 (PSI-4)。意向治疗分析表明，与接受常规治疗的男孩相比，通过远程医疗接受 FCT 的男孩的 ABC-C 易怒分量表（我们的主要结果衡量指标）得分显着降低（p < .001，Cohen 的 d = 0.65）。对接受治疗的人进行的会期观察表明，问题行为水平比基线下降了 91%。FCT 治疗后，与儿童行为问题相关的育儿压力水平也较低，护理人员报告说干预是可以接受的。这些发现支持支持远程医疗的 FCT 作为一个框架，用于扩大对 FXS 儿童问题行为的行为治疗的可及性。通过远程医疗扩大行为分析治疗的提供也有可能降低医疗保健成本，改善儿童和家庭的生活质量，并在更广泛的神经发育障碍人群中促进问题行为的治疗。ClinicalTrials.gov，NCT03510156。2018 年 4 月 27 日注册 通过远程医疗扩大行为分析治疗的提供也有可能降低医疗保健成本，改善儿童和家庭的生活质量，并在更广泛的神经发育障碍人群中促进问题行为的治疗。ClinicalTrials.gov，NCT03510156。2018 年 4 月 27 日注册 通过远程医疗扩大行为分析治疗的提供也有可能降低医疗保健成本，改善儿童和家庭的生活质量，并在更广泛的神经发育障碍人群中促进问题行为的治疗。ClinicalTrials.gov，NCT03510156。2018 年 4 月 27 日注册