Longitudinal outcomes of hearing-impaired children: early vs later intervention

项目总结/摘要 永久性儿童听力损失发生在1-2/1000活产婴儿中， 儿童的生命带来高昂的社会成本。新生儿听力筛查（UNHS）可帮助 听力损失的检测和干预，目的是改善人群的长期结果 水平尽管UNHS被广泛采用，但仍然存在严重的证据缺口。早期的有效性 改善长期结果的干预措施是不确定的，成本效益未经证实， 设备适配对单侧听力损失儿童结局的影响尚不清楚。 通过利用澳大利亚独特的环境，儿童获得统一的听力服务， 从一个单一的供应商（澳大利亚听证会，AH），但有差别访问UNHS在一个狭窄的 时间窗口，研究小组开始了听力儿童的纵向结果 2005年的LOCHI研究。通过NIH的支持，一个基于人群的450名儿童队列， 双侧听力损失（53%在6个月龄前安装助听器）已入组， 对10年的成果进行了前瞻性衡量。5年的数据显示， 儿童接受助听器装配或人工耳蜗植入，更好的语言效果。好处 对于那些听力损失更严重的人来说，这些早期的好处会延伸到以后吗？ 教育程度、心理健康和生活质量？早期干预的成本- 长期有效？此外，早期干预是否能改善出生时患有 单侧听力损失？作为通过UNHS检测到的非目标条件， 因为缺乏证据。在美国国立卫生研究院的支持下， （CUHL）研究入组了179名儿童，其中约50%的儿童在术后被随机分配到装置装配组。 诊断.该应用程序利用了LOCHI和CUHL队列。目标是：（1） 通过测量LOCHI队列的结局来确定早期干预的长期有效性 （2）通过使用以下方法进行经济评估来确定成本效益： 从LOCHI队列中收集的结局和医疗资源使用数据;以及（3） 确定助听器的安装和使用如何影响单侧听力障碍儿童的语言结果 通过评估3岁和5岁时的CUHL队列来评估听力损失。所有数据和一系列预测因素 将在多元回归分析和结构方程模型中进行评估。作为研究机构， 我们将通过AH服务提供网络保持联系，以尽量减少后续损失。 实现这些目标将a）提供急需的证据来指导临床最佳实践 儿童听力损失的全方位管理，包括单侧听力损失;以及 B）展示联合国人类住区规划署和早期干预的成本效益，以指导政策决定。

PROJECT SUMMARY/ ABSTRACT Permanent childhood hearing loss, which occurs in 1-2/1000 live-births, has negative impacts on children's lives incurring high societal costs. Universal newborn hearing screening (UNHS) enables early detection of hearing loss and intervention, with the goal of improving long-term outcomes at a population level. Despite widespread adoption of UNHS, serious evidence gaps remain. The effectiveness of early intervention for improving long-term outcomes is uncertain, the cost-effectiveness is unproven, and the impact of device fitting on outcomes of children with unilateral hearing loss is unknown. By capitalizing on a unique environment in Australia where children receive uniform hearing services from a single provider (Australian Hearing, AH), but had differential access to UNHS during a narrow time window, the research team commenced the Longitudinal Outcomes of Children with Hearing Impairment (LOCHI) study in 2005. Through NIH support, a population-based cohort of 450 children with bilateral hearing loss (53% fitted with hearing aids before age 6 months) have been enrolled, and their outcomes have been measured prospectively over 10 years. The 5-year data revealed that the earlier children received hearing-aid fitting or cochlear implantation, the better the language outcomes. Benefits were greater for those with more severe hearing loss. Would these early benefits extend to later educational attainment, psychosocial well-being, and quality of life? Is early intervention cost- effectiveness in the long term? Further, does early intervention improve outcomes of children born with unilateral hearing loss? As a non-target condition detected via UNHS, there is clinical equipoise about management because evidence is lacking. Supported by NIH, the Children with Unilateral Hearing Loss (CUHL) study enrolled 179 children, ~50% of whom were randomly assigned to fitting of devices after diagnosis. This application draws on the assembled LOCHI and CUHL cohorts. The Aims are to: (1) determine the long-term effectiveness of early intervention by measuring outcomes of the LOCHI cohort at 16 years of age; (2) determine the cost-effectiveness by performing an economic evaluation using data on outcomes and healthcare resource use to be collected from the LOCHI cohort; and (3) determine how fitting and use of hearing device influence language outcomes of children with unilateral hearing loss by evaluating the CUHL cohort at 3 and 5 years of age. All data and a range of predictors will be evaluated in multiple regression analyses and structural equation modelling. As the research arm of AH, we will minimise loss to follow-up by maintaining contact via the AH service provision network. Accomplishing the aims will a) provide the much-needed evidence to guide clinical best-practice management of children across the entire spectrum of hearing loss, including unilateral hearing loss; and b) demonstrate the cost-effectiveness of UNHS and early intervention to guide policy decisions.