Lucas Aaron, Sinha Anushua, Fowler Karen B, Mladsi Deirdre, Barnett Christine, Samant Salome, Gibson Laura
1RTI Health Solutions, 3040 Cornwallis Road, PO Box 12194, Research Triangle Park, NC 27709-2194 USA.
2Center for Observational and Real-World Evidence (CORE), Merck & Co., Inc., Kenilworth, NJ USA.
Cost Eff Resour Alloc. 2019 Oct 3;17:21. doi: 10.1186/s12962-019-0189-0. eCollection 2019.
In the United States (US), congenital cytomegalovirus infection (cCMVi) is a major cause of permanent disabilities and the most common etiology of non-genetic sensorineural hearing loss. Evaluations of prevention strategies will require estimates of the economic implications of cCMVi. We aimed to develop a conceptual framework to characterize the lifetime economic burden of cCMVi in the US and to use that framework to identify data gaps.
Direct health care, direct non-health care, indirect, and intangible costs associated with cCMVi were considered. An initial framework was constructed based on a targeted literature review, then validated and refined after consultation with experts. Published costs were identified and used to populate the framework. Data gaps were identified.
The framework was constructed as a chance tree, categorizing clinical event occurrence to form patient profiles associated with distinct economic trajectories. The distribution and magnitude of costs varied by patient life stage, cCMVi diagnosis, severity of impairment, and developmental delays/disabilities. Published studies could not fully populate the framework. The literature best characterized direct health care costs associated with the birth period. Gaps existed for direct non-health care, indirect, and intangible costs, as well as health care costs associated with adult patients and those severely impaired.
Data gaps exist concerning the lifetime economic burden of cCMVi in the US. The conceptual framework provides the basis for a research agenda to address these gaps. Understanding the full lifetime economic burden of cCMVi would inform clinicians, researchers, and policymakers, when assessing the value of cCMVi interventions.
在美国，先天性巨细胞病毒感染（cCMVi）是导致永久性残疾的主要原因，也是非遗传性感音神经性听力损失最常见的病因。对预防策略的评估将需要对cCMVi的经济影响进行估计。我们旨在建立一个概念框架，以描述美国cCMVi的终生经济负担，并利用该框架识别数据缺口。
考虑了与cCMVi相关的直接医疗保健、直接非医疗保健、间接和无形费用。基于有针对性的文献综述构建了一个初始框架，然后在与专家协商后进行验证和完善。确定已发表的费用并用于填充该框架。识别数据缺口。
该框架构建为一个机会树，对临床事件的发生进行分类，以形成与不同经济轨迹相关的患者概况。费用的分布和规模因患者生命阶段、cCMVi诊断、损伤严重程度以及发育迟缓/残疾情况而异。已发表的研究无法完全填充该框架。文献对与出生期相关的直接医疗保健费用描述得最为详尽。在直接非医疗保健、间接和无形费用以及与成年患者和严重受损患者相关的医疗保健费用方面存在缺口。
在美国，关于cCMVi终生经济负担的数据存在缺口。该概念框架为解决这些缺口的研究议程提供了基础。了解cCMVi的完整终生经济负担将为临床医生、研究人员和政策制定者在评估cCMVi干预措施的价值时提供参考。
