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对二叶式主动脉瓣患者一级亲属进行筛查的成本效益分析。

Cost-effectiveness analysis of screening for first-degree relatives of patients with bicuspid aortic valve.

作者信息

Tessler Idit, Leshno Moshe, Shmueli Amir, Shpitzen Shoshana, Durst Ronen, Gilon Dan

机构信息

Braun School of Public Health and Community Medicine, Faculty of Medicine, The Hebrew University, Jerusalem, Israel.

Heart Institute, Hadassah Medical Center, Jerusalem 91120, Israel.

出版信息

Eur Heart J Qual Care Clin Outcomes. 2021 Sep 16;7(5):447-457. doi: 10.1093/ehjqcco/qcab047.

Abstract

AIMS

Bicuspid aortic valve (BAV) is the commonest congenital heart valve malformation, and is associated with life-threatening complications. Given the high heritability index of BAV, many experts recommend echocardiography screening for first-degree relatives (FDRs) of an index case. Here, we aim to evaluate the cost-effectiveness of such cascade screening for BAV.

METHODS AND RESULTS

Using a decision-analytic model, we performed a cost-effectiveness analysis of echocardiographic screening for FDRs of a BAV index case. Data on BAV probabilities and complications among FDRs were derived from our institution's BAV familial cohort and from the literature on population-based BAV cohorts with long-term follow-up. Health gain was measured as quality-adjusted life years (QALYs). Cost inputs were based on list prices and literature data. One-way and probabilistic sensitivity analyses were performed to account for uncertainty in the model's variables. Screening of FDRs was found to be the dominant strategy, being more effective and less costly than no screening, with savings of €644 and gains of 0.3 QALY. Results were sensitive throughout the range of the main model's variables, including the full range of reported BAV rates among FDRs across the literature. A gradual decrease of the incremental effect was found with the increase in screening age.

CONCLUSION

This economic evaluation model found that echocardiographic screening of FDRs of a BAV index case is not only clinically important but also cost-effective and cost-saving. Sensitivity analysis supported the model's robustness, suggesting its generalization.

摘要

目的

二叶式主动脉瓣(BAV)是最常见的先天性心脏瓣膜畸形,并伴有危及生命的并发症。鉴于BAV的高遗传指数,许多专家建议对索引病例的一级亲属(FDR)进行超声心动图筛查。在此,我们旨在评估这种针对BAV的级联筛查的成本效益。

方法与结果

我们使用决策分析模型,对BAV索引病例的FDR进行超声心动图筛查的成本效益进行了分析。FDR中BAV概率和并发症的数据来自我们机构的BAV家族队列以及关于长期随访的基于人群的BAV队列的文献。健康收益以质量调整生命年(QALY)衡量。成本投入基于标价和文献数据。进行了单向和概率敏感性分析,以考虑模型变量中的不确定性。发现对FDR进行筛查是主要策略,比不进行筛查更有效且成本更低,节省了644欧元,获得了0.3个QALY。结果在主要模型变量的整个范围内都很敏感,包括文献中报道的FDR中BAV发生率的整个范围。随着筛查年龄的增加,增量效应逐渐降低。

结论

该经济评估模型发现,对BAV索引病例的FDR进行超声心动图筛查不仅在临床上很重要,而且具有成本效益且节省成本。敏感性分析支持了该模型的稳健性,表明其具有推广性。

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