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都有长期的负面影响，包括社会发展、教育和就业机会，以及情感健康和心理健康（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得到改善。家长与孩子一起（PACT）是一个由家长提供的早期语言教学计划，为典型的发展中学前儿童在语言延迟的风险。先前的项目（Burgoyne等人，2018年）表明，PACT导致儿童语言和早期识字技能的改善。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。来自专家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有潜力支持唐氏综合症儿童的语言学习，并建议如何调整该方案，以提高其对这类儿童的有效性。本研究的目的是调整和评估PACT作为唐氏综合征幼儿的父母提供的语言干预。将通过两个阶段的工作实现这一目标，其目标如下：（1）开展试点研究，以确定和评估对PACT方案的调整。在项目的第一年，我们将与一小群患有唐氏综合症的儿童（5名儿童; 3-6岁）的父母密切合作，探讨他们使用PACT的经验，并确定对该计划的调整，以提高其有效性。家长将被要求参加两个周期的试点和反馈。在第一个周期中，父母将为孩子提供为期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，交付观察，简单的调查和焦点小组。我们将利用这些信息对为期5周的PACT计划进行修改，家长将在第二个试点周期中提供。第2周期的家长反馈将评估我们所做的改变，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)进行一项可行性随机对照试验，以评估干预的可行性，并探索该计划在支持唐氏综合征幼儿语言和识字发展方面的潜力。我们将招募24名患有唐氏综合症的儿童（3-6岁）及其父母参加。儿童将被随机分配到PACT+组或等待对照组（在项目结束时接受干预）。PACT+小组将接受培训，每天（每天20分钟;每周5天）为他们的孩子提供PACT+，持续30周。我们将通过评估儿童语言和早期识字能力、衡量父母福祉、定性调查和访谈以及监测数据收集数据，以探讨干预的可行性（包括招募和保留家庭，以及干预的坚持性和可接受性）以及成本和效益。将在干预前、30周干预计划后立即和干预结束后6个月收集数据。这项研究有可能对我们理解如何支持唐氏综合症幼儿的父母在家中发展孩子的语言发展，并改善该组的语言成果产生重大影响。

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.