O'Toole Ciara, Lee Alice S-Y, Gibbon Fiona E, van Bysterveldt Anne K, Hart Nicola J
Department of Speech and Hearing Sciences, University College Cork, Brookfield Health Sciences Complex, College Road, Cork, Ireland.
Cochrane Database Syst Rev. 2018 Oct 15;10(10):CD012089. doi: 10.1002/14651858.CD012089.pub2.
Communication and language development are areas of particular weakness for young children with Down syndrome. Caregivers' interaction with children influences language development, so many early interventions involve training parents how best to respond to their children and provide appropriate language stimulation. Thus, these interventions are mediated through parents, who in turn are trained and coached in the implementation of interventions by clinicians. As the interventions involve a considerable commitment from clinicians and families, we undertook this review to synthesise the evidence of their effectiveness.
To assess the effects of parent-mediated interventions for improving communication and language development in young children with Down syndrome. Other outcomes are parental behaviour and responsivity, parental stress and satisfaction, and children's non-verbal means of communicating, socialisation and behaviour.
In January 2018 we searched CENTRAL, MEDLINE, Embase and 14 other databases. We also searched three trials registers, checked the reference lists of relevant reports identified by the electronic searches, searched the websites of professional organizations, and contacted their staff and other researchers working in the field to identify other relevant published, unpublished and ongoing studies.
We included randomised controlled trials (RCTs) and quasi-RCTs that compared parent-mediated interventions designed to improve communication and language versus teaching/treatment as usual (TAU) or no treatment or delayed (wait-listed) treatment, in children with Down syndrome aged between birth and six years. We included studies delivering the parent-mediated intervention in conjunction with a clinician-mediated intervention, as long as the intervention group was the only group to receive the former and both groups received the latter.
We used standard Cochrane methodological procedures for data collection and analysis.
We included three studies involving 45 children aged between 29 months and six years with Down syndrome. Two studies compared parent-mediated interventions versus TAU; the third compared a parent-mediated plus clinician-mediated intervention versus a clinician-mediated intervention alone. Treatment duration varied from 12 weeks to six months. One study provided nine group sessions and four individualised home-based sessions over a 13-week period. Another study provided weekly, individual clinic-based or home-based sessions lasting 1.5 to 2 hours, over a six-month period. The third study provided one 2- to 3-hour group session followed by bi-weekly, individual clinic-based sessions plus once-weekly home-based sessions for 12 weeks. Because of the different study designs and outcome measures used, we were unable to conduct a meta-analysis.We judged all three studies to be at high risk of bias in relation to blinding of participants (not possible due to the nature of the intervention) and blinding of outcome assessors, and at an unclear risk of bias for allocation concealment. We judged one study to be at unclear risk of selection bias, as authors did not report the methods used to generate the random sequence; at high risk of reporting bias, as they did not report on one assessed outcome; and at high risk of detection bias, as the control group had a cointervention and only parents in the intervention group were made aware of the target words for their children. The sample sizes of each included study were very small, meaning that they are unlikely to be representative of the target population.The findings from the three included studies were inconsistent. Two studies found no differences in expressive or receptive language abilities between the groups, whether measured by direct assessment or parent reports. However, they did find that children in the intervention group could use more targeted vocabulary items or utterances with language targets in certain contexts postintervention, compared to those in the control group; this was not maintained 12 months later. The third study found gains for the intervention group on total-language measures immediately postintervention.One study did not find any differences in parental stress scores between the groups at any time point up to 12 months postintervention. All three studies noted differences in most measures of how the parents talked to and interacted with their children postintervention, and in one study most strategies were maintained in the intervention group at 12 months postintervention. No study reported evidence of language attrition following the intervention in either group, while one study found positive outcomes on children's socialisation skills in the intervention group. One study looked at adherence to the treatment through attendance data, finding that mothers in the intervention group attended seven out of nine group sessions and were present for four home visits. No study measured parental use of the strategies outside of the intervention sessions.A grant from the Hospital for Sick Children Foundation (Toronto, Ontario, Canada) funded one study. Another received partial funding from the National Institute of Child Health and Human Development and the Department of Education in the USA. The remaining study did not specify any funding sources.In light of the serious limitations in methodology, and the small number of studies included, we considered the overall quality of the evidence, as assessed by GRADE, to be very low. This means that we have very little confidence in the results, and further research is very likely to have an important impact on our confidence in the estimate of treatment effect.
AUTHORS' CONCLUSIONS: There is currently insufficient evidence to determine the effects of parent-mediated interventions for improving the language and communication of children with Down syndrome. We found only three small studies of very low quality. This review highlights the need for well-designed studies, including RCTs, to evaluate the effectiveness of parent-mediated interventions. Trials should use valid, reliable and similar measures of language development, and they should include measures of secondary outcomes more distal to the intervention, such as family well-being. Treatment fidelity, in particular parental dosage of the intervention outside of prescribed sessions, also needs to be documented.
沟通和语言发展是唐氏综合征幼儿特别薄弱的领域。照顾者与孩子的互动会影响语言发展,因此许多早期干预措施包括培训父母如何最好地回应孩子并提供适当的语言刺激。因此,这些干预是通过父母来实施的,而父母又会在临床医生的培训和指导下实施干预措施。由于这些干预措施需要临床医生和家庭投入大量精力,我们进行了这项综述,以综合其有效性的证据。
评估父母介导的干预措施对改善唐氏综合征幼儿沟通和语言发展的效果。其他结果包括父母的行为和反应性、父母的压力和满意度,以及孩子的非语言沟通方式、社交能力和行为。
2018年1月,我们检索了Cochrane中心对照试验注册库(CENTRAL)、医学期刊数据库(MEDLINE)、荷兰医学文摘数据库(Embase)和其他14个数据库。我们还检索了三个试验注册库,检查了电子检索所识别的相关报告的参考文献列表,搜索了专业组织的网站,并联系了他们的工作人员以及该领域的其他研究人员,以识别其他相关的已发表、未发表和正在进行的研究。
我们纳入了随机对照试验(RCT)和半随机对照试验,这些试验比较了旨在改善沟通和语言的父母介导干预措施与常规教学/治疗(TAU)或无治疗或延迟(等待名单)治疗,对象为出生至6岁的唐氏综合征儿童。我们纳入了将父母介导干预措施与临床医生介导干预措施结合实施的研究,只要干预组是唯一接受前者的组,且两组均接受后者。
我们采用Cochrane标准方法程序进行数据收集和分析。
我们纳入了三项研究,涉及45名年龄在29个月至6岁之间的唐氏综合征儿童。两项研究比较了父母介导干预措施与TAU;第三项研究比较了父母介导加临床医生介导干预措施与仅临床医生介导干预措施。治疗持续时间从12周至6个月不等。一项研究在13周内提供了9次小组课程和4次个性化的家庭课程。另一项研究在6个月内每周提供1.5至2小时的基于诊所或家庭的个人课程。第三项研究提供了一次2至3小时的小组课程,随后在12周内每两周提供一次基于诊所的个人课程以及每周一次的家庭课程。由于所使用的研究设计和结局测量方法不同,我们无法进行荟萃分析。我们判断所有三项研究在参与者盲法(由于干预措施的性质无法实现)和结局评估者盲法方面存在高偏倚风险,在分配隐藏方面存在不明确的偏倚风险。我们判断一项研究在选择偏倚方面存在不明确风险,因为作者未报告用于生成随机序列的方法;在报告偏倚方面存在高风险,因为他们未报告一项评估结局;在检测偏倚方面存在高风险,则是因为对照组有一项联合干预措施,且只有干预组的父母知晓其孩子的目标词汇。每项纳入研究的样本量都非常小,这意味着它们不太可能代表目标人群。三项纳入研究的结果不一致。两项研究发现,无论是通过直接评估还是父母报告,两组在表达性或接受性语言能力方面均无差异。然而,他们确实发现,与对照组相比,干预组的儿童在干预后能够在某些情境中使用更多有针对性的词汇项目或带有语言目标的话语;但12个月后这种情况并未持续。第三项研究发现干预组在干预后立即在总语言测量方面有所收获。一项研究发现,在干预后长达12个月的任何时间点,两组之间的父母压力得分均无差异。所有三项研究均指出,干预后父母与孩子交谈和互动的大多数测量指标存在差异,并且在一项研究中,干预组在干预后12个月时大多数策略仍得以维持。没有研究报告两组在干预后语言消退的证据,而一项研究发现干预组在儿童社交技能方面有积极结果。一项研究通过出勤数据查看了对治疗的依从性,发现干预组的母亲参加了9次小组课程中的7次,并接受了4次家访。没有研究测量父母在干预课程之外对策略的使用情况。一项研究由加拿大安大略省多伦多市病童医院基金会资助。另一项研究获得了美国国立儿童健康与人类发展研究所和教育部的部分资助。其余研究未指明任何资金来源。鉴于方法学上的严重局限性以及纳入研究数量较少,我们认为根据GRADE评估的证据总体质量非常低。这意味着我们对结果的信心非常小,进一步的研究很可能会对我们对治疗效果估计的信心产生重要影响。
目前尚无足够证据确定父母介导的干预措施对改善唐氏综合征儿童语言和沟通的效果。我们仅发现三项质量极低的小型研究。本综述强调需要进行精心设计的研究,包括随机对照试验,以评估父母介导干预措施的有效性。试验应使用有效、可靠且相似的语言发展测量方法,并且应包括对干预更具远期影响的次要结局的测量,如家庭幸福感。还需要记录治疗的保真度,特别是父母在规定课程之外对干预措施的实施情况。