Macquarie University Centre for the Health Economy, Macquarie Business School, Macquarie University, NSW, Australia.
Department of Economics, Macquarie University, NSW, Australia.
Ear Hear. 2022 May/Jun;43(3):972-983. doi: 10.1097/AUD.0000000000001153.
Hearing loss is one of the most prevalent congenital disorders among children. Many countries have implemented universal newborn hearing screening (UNHS) for the early diagnosis and treatment of hearing loss. Despite widespread implementation, the value for money of UNHS is unclear due to lack of cost and outcomes data from rigorous study designs. The objective of this research is to conduct a within-study cost-effectiveness analysis of UNHS compared with targeted screening (targeting children with risk factors of hearing loss) from the Australian healthcare system perspective. This evaluation is the first economic evaluation to assess the cost-effectiveness of UNHS compared to targeted screening using real-world data from a natural experiment.
The evaluation assumed the Australian healthcare system perspective and considered a time horizon of 5 years. Utilities were estimated using responses to the Health Utilities Index Mark III. Screening costs were estimated based on the Victorian Infant Hearing Screening Program. Ongoing costs were estimated based on administrative data, while external data sources were used to estimate costs related to hearing services. Missing data were handled using the multiple imputation method. Outcome measures included quality-adjusted life years (QALYs) and four language and communication-related outcomes: Peabody Picture Vocabulary Test, Wechsler Nonverbal Scale of Ability, Progressive Achievement Test, and comprehensive, expressive, and total language scores based on the Preschool Language Scale.
On average, the UNHS cost an extra Australian dollar (A$)22,000 per diagnosed child and was associated with 0.45 more QALYs per diagnosed child compared with targeted screening to 5 years, resulting in an incremental cost-effectiveness ratio (ICER) of A$48,000 per QALY gained. The ICERs for language outcomes lay between A$3,900 (for expressive language score) and A$83,500 per one-point improvement in language score (for Wechsler Nonverbal Scale of Ability). UNHS had a 69% probability of being more cost-effective compared to targeted screening at a willingness to pay threshold of A$60,000 per QALY gained. ICERs were most sensitive to the screening costs.
The evaluation demonstrated the usefulness of a within-study economic evaluation to understand the value for money of the UNHS program in the Australian context. Findings from this evaluation suggested that screening costs were the key driver of cost-effectiveness results. Most outcomes were not significantly different between UNHS and targeted screening groups. The ICER may be overestimated due to the short follow-up period. Further research is warranted to include long-term resource use and outcome data, late diagnosis, transition and remission between severity levels, and timing of diagnosis and treatment.
听力损失是儿童中最常见的先天性疾病之一。许多国家已经实施了新生儿普遍听力筛查（UNHS），以早期诊断和治疗听力损失。尽管已经广泛实施，但由于缺乏严格研究设计的成本和结果数据，UNHS 的性价比尚不清楚。本研究的目的是从澳大利亚医疗保健系统的角度，对 UNHS 与目标筛查（针对有听力损失风险因素的儿童）进行基于研究的成本效益分析。这是首次使用自然实验的真实世界数据来评估 UNHS 与目标筛查相比的成本效益的经济评估。
该评估采用澳大利亚医疗保健系统的观点，并考虑了 5 年的时间范围。效用值使用健康效用指数 III 量表的回答进行估算。筛查成本基于维多利亚婴儿听力筛查计划进行估算。持续成本根据行政数据进行估算，而外部数据源用于估算与听力服务相关的成本。使用多重插补法处理缺失数据。结果指标包括质量调整生命年（QALYs）和四个语言和沟通相关结果：皮博迪图片词汇测验、韦氏非言语能力量表、进步成就测验以及基于学前语言量表的全面、表达和总语言分数。
平均而言，与目标筛查相比，UNHS 为每诊断出的儿童额外花费 22,000 澳元（A$），并与每诊断出的儿童多获得 0.45 个 QALY，至 5 年时，增量成本效益比（ICER）为每获得一个 QALY 增加 A$48,000。语言结果的 ICER 值在 A$3,900（表达语言分数）和 A$83,500（韦氏非言语能力量表）之间每提高一分语言分数。在愿意支付每获得一个 QALY 增加 A$60,000 的阈值下，UNHS 比目标筛查更具成本效益的可能性为 69%。ICER 对筛查成本最为敏感。
本评估展示了基于研究的经济评估在理解澳大利亚 UNHS 计划性价比方面的实用性。评估结果表明，筛查成本是成本效益结果的关键驱动因素。UNHS 组和目标筛查组的大多数结果没有显著差异。由于随访时间较短，ICER 可能被高估。需要进一步研究以纳入长期资源使用和结果数据、迟发性诊断、严重程度水平之间的过渡和缓解，以及诊断和治疗的时间。
