Abstract：
OBJECTIVE: Diagnosis rate of familial hypercholesterolemia (FH) remained less than 10 % globally and the economic evaluation results of different FH screening strategies varied. This study aimed to systematically review the methodology and results of cost effectiveness analysis (CEA) of FH screening, which will provide evidence support for health-related decision-making.
METHODS: The Medline/PubMed, Embase, Cochrane Library, Web of science, National Health Service Economic Evaluation Database (NHSEED) and CEA Registry databases were electronically searched to collect full economic evaluation from the establishment of the databases to June 30, 2022. The quality of included studies was evaluated by the Consolidated Health Economic Evaluation Reporting Standards statement 2022 (CHEERS 2022) checklist.
RESULTS: Among 232 retrieved studies, 18 economic evaluations were included and all of them are from developed countries, with an average quality score of 0.73. The decision tree model and/or Markov model were constructed by thirteen articles (72 %). Twelve studies (67 %) adopted the healthcare perspective and the lifetime horizon to compare the costs and health outcome of different screening strategies. The results of eight studies indicated that cascade screening was a cost-effective strategy compared with no screening, which was more pronounced in younger adults. Universal screening in young adults aged 16 years or 18-40 years (n=3) and in children aged 1-2 years combined with reverse cascade screening (n=3) are both cost-effective. The probability of being cost-effective for cascade screening (n=6) and universal screening (n=1) of young aged 18-40 years were greater than 95 %.
CONCLUSIONS: Our review demonstrated the economic advantages of cascade screening, universal screening of young adults, and universal screening of newborns combined with reverse cascade screening. Further health economic evaluation is needed in children and in low- and middle-income countries.
METHODS: The Medline/PubMed, Embase, Cochrane Library, Web of science, National Health Service Economic Evaluation Database (NHSEED) and CEA Registry databases were electronically searched to collect full economic evaluation from the establishment of the databases to June 30, 2022. The quality of included studies was evaluated by the Consolidated Health Economic Evaluation Reporting Standards statement 2022 (CHEERS 2022) checklist.
RESULTS: Among 232 retrieved studies, 18 economic evaluations were included and all of them are from developed countries, with an average quality score of 0.73. The decision tree model and/or Markov model were constructed by thirteen articles (72 %). Twelve studies (67 %) adopted the healthcare perspective and the lifetime horizon to compare the costs and health outcome of different screening strategies. The results of eight studies indicated that cascade screening was a cost-effective strategy compared with no screening, which was more pronounced in younger adults. Universal screening in young adults aged 16 years or 18-40 years (n=3) and in children aged 1-2 years combined with reverse cascade screening (n=3) are both cost-effective. The probability of being cost-effective for cascade screening (n=6) and universal screening (n=1) of young aged 18-40 years were greater than 95 %.
CONCLUSIONS: Our review demonstrated the economic advantages of cascade screening, universal screening of young adults, and universal screening of newborns combined with reverse cascade screening. Further health economic evaluation is needed in children and in low- and middle-income countries.
摘要：
目的：全球家族性高胆固醇血症(FH)诊断率仍低于10%，不同FH筛查策略的经济学评价结果各不相同。本研究旨在系统回顾FH筛查的成本效益分析(CEA)的方法和结果。这将为健康相关决策提供证据支持。
方法：Medline/PubMed，Embase,科克伦图书馆,WebofScience,对国家卫生服务经济评价数据库（NHSEED）和CEA注册数据库进行了电子搜索，以收集从数据库建立到2022年6月30日的完整经济评价。纳入研究的质量通过2022年综合卫生经济评估报告标准声明（CHEERS2022）清单进行评估。
结果：在检索到的232项研究中，包括18项经济评估，所有这些评估都来自发达国家，平均质量评分为0.73。决策树模型和/或马尔可夫模型由13篇文章(72%)构建。12项研究（67％）采用了医疗保健观点和生命周期来比较不同筛查策略的成本和健康结果。八项研究的结果表明，与不进行筛查相比，级联筛查是一种具有成本效益的策略，这在年轻人中更为明显。16岁或18-40岁的年轻人（n=3）和1-2岁的儿童联合反向级联筛查（n=3）的普遍筛查都具有成本效益。对于18-40岁的年轻人进行级联筛查（n=6）和通用筛查（n=1）具有成本效益的可能性大于95％。
结论：我们的综述证明了级联筛查的经济优势，对年轻人进行普遍筛查，和新生儿的普遍筛查结合反向级联筛查。需要对儿童以及低收入和中等收入国家进行进一步的卫生经济评估。
方法：Medline/PubMed，Embase,科克伦图书馆,WebofScience,对国家卫生服务经济评价数据库（NHSEED）和CEA注册数据库进行了电子搜索，以收集从数据库建立到2022年6月30日的完整经济评价。纳入研究的质量通过2022年综合卫生经济评估报告标准声明（CHEERS2022）清单进行评估。
结果：在检索到的232项研究中，包括18项经济评估，所有这些评估都来自发达国家，平均质量评分为0.73。决策树模型和/或马尔可夫模型由13篇文章(72%)构建。12项研究（67％）采用了医疗保健观点和生命周期来比较不同筛查策略的成本和健康结果。八项研究的结果表明，与不进行筛查相比，级联筛查是一种具有成本效益的策略，这在年轻人中更为明显。16岁或18-40岁的年轻人（n=3）和1-2岁的儿童联合反向级联筛查（n=3）的普遍筛查都具有成本效益。对于18-40岁的年轻人进行级联筛查（n=6）和通用筛查（n=1）具有成本效益的可能性大于95％。
结论：我们的综述证明了级联筛查的经济优势，对年轻人进行普遍筛查，和新生儿的普遍筛查结合反向级联筛查。需要对儿童以及低收入和中等收入国家进行进一步的卫生经济评估。
