Parent-Delivered Early Language Intervention for Children with Down Syndrome: A Randomised Controlled Feasibility Trial

唐氏综合症是导致智力残疾的最常见的遗传原因，在英国每年影响700-800名婴儿(Wu&Morris，2013)。患有唐氏综合症的儿童在发展语言技能方面存在困难。这些困难对他们生活的方方面面都有长期的负面影响，包括社会发展、教育和就业机会、情感健康和精神健康(世界卫生组织，2007年)。这些方面都有可能通过有针对性的早期语言干预而得到改善。家长和孩子在一起(PACT)是一项由父母提供的早期语言教学计划，适用于有语言延迟风险的典型发育的学龄前儿童。先前的一个项目(Burgoyne等人，2018年)表明，PACT有助于提高儿童的语言和早期识字技能。PACT包含许多支持唐氏综合症儿童学习的功能，可能对这一群体有用。来自专业言语和语言治疗师以及唐氏综合症儿童家长的非正式反馈表明，PACT有可能支持唐氏综合症儿童的语言学习，并就如何调整该方案以提高对这一儿童群体的有效性提出建议。目前这项研究的目的是适应和评估PACT作为一种父母递送的语言干预措施来治疗唐氏综合症儿童。这一目标将通过以下两个阶段的工作来实现：1)开展一项试点研究，以确定和评估适应《公约》方案的情况。在项目的第一年，我们将与一小群有唐氏综合症儿童(5名儿童，3-6岁)的父母密切合作，探索他们使用PACT的经验，并确定对该计划的调整，以提高其有效性。家长将被要求参加试点和反馈两个周期。在第一个周期中，父母将向他们的孩子交付5周的原始PACT计划。我们将使用记录表、分娩观察、简单调查和焦点小组收集关于他们的经验的数据，包括可以改进的干预方面。我们将利用这些信息对另外5周的PACT计划进行修改，家长将在第二个试行周期中提供这些计划。来自第二周期的家长反馈将评估我们所做的更改，并确定对该计划的任何进一步调整。这一进程将产生一个完全适应唐氏综合症父母和幼儿需要的方案(我们称之为PACT+)，供第二阶段使用。2)进行可行性随机对照试验，以评估干预措施的可行性，并探索该计划在支持唐氏综合症幼儿语言和识字发展方面的潜力。我们将招募24名唐氏综合征儿童(3-6岁)及其父母参加。儿童将被随机分配到PACT+组或等待控制组(将在项目结束时接受干预)。PACT+组将接受培训，每天(每天20分钟；每周5天)为他们的孩子提供30周以上的PACT+。我们将使用儿童语言和早期识字评估、父母幸福感测量、定性调查和访谈以及监测数据收集数据，以探索干预措施的可行性(包括招募和保留家庭，以及干预措施的坚持和可接受程度)以及成本和收益。数据将在干预前、30周干预计划后立即和干预结束后6个月收集。这项研究可能会对我们理解如何支持唐氏综合症儿童的父母在家中发展他们的孩子的语言发展，并改善这一群体的语言结果产生重大影响。

Down syndrome is the most common genetic cause of intellectual disability, affecting 700-800 babies annually in the UK (Wu & Morris, 2013). Children with Down syndrome have difficulties developing language skills. These difficulties have long term negative consequences for all aspects of their lives including social development, education and employment opportunities, and emotional wellbeing and mental health (World Health Organisation, 2007). These aspects all have the potential to be improved through targeted early language intervention. Parents and Children Together (PACT) is a parent-delivered early language teaching programme for typically-developing pre-school children at risk of language delays. A previous project (Burgoyne et al., 2018) showed that PACT leads to improvements in children's language and early literacy skills. PACT contains many features which support learning for children with Down syndrome, and could be useful for this group. Informal feedback from specialist speech and language therapists and parents of children with Down syndrome suggests that PACT has potential to support language learning for children with Down syndrome and suggested ways the programme could be adapted to enhance its effectiveness for this group of children. The aim of the current study is to adapt and evaluate PACT as a parent-delivered language intervention for young children with Down syndrome. This aim will be met through two phases of work with the following objectives: 1) To carry out a pilot study to identify and evaluate adaptations to the PACT programme. Over the first year of the project, we will work closely with a small group of parents who have a child with Down syndrome (5 children; aged 3-6 years) to explore their experiences of using PACT, and identify adaptations to the programme to enhance its effectiveness. Parents will be asked to take part in two cycles of piloting and feedback. In the first cycle, parents will deliver 5 weeks of the original PACT programme to their child. We will collect data on their experiences, including aspects of the intervention that could be improved, using record forms, observations of delivery, simple surveys, and a focus group. We will use this information to make changes to a further 5-weeks of the PACT programme, which parents will deliver in the second cycle of piloting. Parent feedback from cycle 2 will evaluate the changes we have made and identify any further adaptations to the programme. This process will lead to a fully adapted programme that is tailored to the needs of parents and young children with Down syndrome (which we call PACT+) for use in the second phase. 2) To conduct a feasibility randomised controlled trial to assess the feasibility of the intervention and explore the programme's potential to support language and literacy development in young children with Down syndrome. We will recruit 24 children with Down syndrome (aged 3-6 years) and their parents to take part. Children will be randomly allocated to either a PACT+ group or to a waiting control group (who will receive intervention at the end of the project). The PACT+ group will be trained to deliver PACT+ every day (20-minutes a day; 5 days a week) to their child over 30-weeks. We will collect data using assessments of child language and early literacy, measures of parent wellbeing, qualitative surveys and interviews, and monitoring data to explore feasibility of the intervention (including recruitment and retention of families, and adherence and acceptability of intervention) and cost and benefits. Data will be collected before intervention, immediately after the 30-week intervention programme, and 6-months after intervention ends. This research has the potential to make a significant difference to our understanding of how to support parents of young children with Down syndrome to develop their child's language development at home, and to improve language outcomes for this group.