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

唐氏综合症是智力残疾最常见的遗传原因，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有困难。这些困难对他们生活的各个方面，包括社会发展、教育和就业机会、情感健康和精神健康，都产生了长期的负面影响（世界卫生组织，2007年）。通过有针对性的早期语言干预，这些方面都有可能得到改善。父母和孩子在一起（PACT）是一个家长提供的早期语言教学项目，针对有语言迟缓风险的学龄前儿童。之前的一个项目（Burgoyne et al., 2018）表明PACT可以提高儿童的语言和早期读写技能。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。来自专业语言治疗师和唐氏综合症儿童父母的非正式反馈表明，PACT有潜力支持唐氏综合症儿童的语言学习，并提出了一些方法，可以调整该计划以提高其对这群儿童的有效性。当前研究的目的是适应和评估PACT作为父母提供的语言干预年幼的唐氏综合症儿童。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，以确定和评价对PACT方案的适应情况。在项目的第一年，我们将与一小群有唐氏综合症孩子的父母（5个孩子，3-6岁）密切合作，探索他们使用PACT的经验，并确定对该计划的适应性，以提高其有效性。家长将被要求参加两个试点和反馈周期。在第一个周期，家长将给孩子提供5周的原始PACT课程。我们将收集他们的经验数据，包括可以改进的干预方面，使用记录表格、执行情况观察、简单调查和焦点小组。我们将利用这些信息对今后5周的PACT项目做出调整，这些家长将在第二轮试点中交付。第二阶段的家长反馈将评估我们所做的改变，并确定对课程的进一步调整。这一进程将导致一个完全适应唐氏综合症父母和幼儿需求的规划（我们称之为PACT+），用于第二阶段。2)开展可行性随机对照试验，评估干预的可行性，探索该项目对唐氏综合征幼儿语言和读写能力发展的支持潜力。我们将招募24名唐氏综合症儿童（3-6岁）及其父母参与。孩子们将被随机分配到PACT+组或等待的对照组（他们将在项目结束时接受干预）。PACT+组将接受培训，在30周内每天（每天20分钟，每周5天）向他们的孩子提供PACT+。我们将使用儿童语言和早期读写能力评估、父母福利测量、定性调查和访谈以及监测数据来收集数据，以探索干预的可行性（包括招募和保留家庭、坚持和接受干预）以及成本和收益。数据将在干预前、30周干预计划结束后立即和干预结束后6个月收集。这项研究有可能对我们的理解产生重大影响，即如何支持唐氏综合症幼儿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。

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.