SynthesisPharmacoEconomics2024
Cost-Effectiveness of Screening Strategies for Familial Hypercholesterolaemia: An Updated Systematic Review.
Synthesis in PharmacoEconomics, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers, 2 of them syntheses that pooled it.
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Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
10 citing papers in PubMed, 2 syntheses or guidelines pooled it, 19 citations in OpenAlex.
- A Critical Systematic Review of Modelling Approaches and Methodologies used in Hyperlipidaemia Economic Evaluations.PharmacoEconomics · 2026Pooled it
- Synthesized economic evidence on the cost-effectiveness of screening familial hypercholesterolemia.Global health research and policy · 2024Pooled it
- Genetic Cascade Screening for Familial Hypercholesterolemia: A Randomized Clinical Trial.JAMA network open · 2026Trial
- Integrating Genetic Counseling in Cardiology Clinics: From Guidelines to Practice.Reviews in cardiovascular medicine · 2026Article
- Observational
- A Cost-Effectiveness Analysis for the Combination of Universal Screening at 9-10 Years Old and Reverse Cascade Screening of Relatives for Familial Hypercholesterolemia in Japan.Journal of atherosclerosis and thrombosis · 2025Article
- Screening for Familial Hypercholesterolemia in Children and its Cost-Effectiveness.Journal of atherosclerosis and thrombosis · 2025Article
- A qualitative study of perceptions of the care pathway for familial hypercholesterolemia: screening, diagnosis, treatment, and family cascade screening.Implementation science communications · 2024Article
- Genetic Testing for Familial Hypercholesterolemia: The Current State of Its Implementation in Canada.CJC open · 2024Article
- The Power of the Pedigree: Cascade Screening in Familial Hypercholesterolemia.JACC. Advances · 2024Article
Corrections and comments
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Authors and funding
10 authors at 8 institutions in 2 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
background
objectiveThis study aimed to systematically synthesise the cost-effectiveness of screening strategies to detect heterozygous familial hypercholesterolemia (FH).
methodsWe searched seven databases from inception to 2 February , 2023, for eligible cost-effective analysis (CEA) that evaluated screening strategies for FH versus the standard care for FH detection. Independent reviewers performed the screening, data extraction and quality evaluation. Cost results were adapted to 2022 US dollars (US$) to facilitate comparisons between studies using the same screening strategies. Cost-effectiveness thresholds were based on the original study criteria.
resultsA total of 21 studies evaluating 62 strategies were included in this review, most of the studies (95%) adopted a healthcare perspective in the base case, and majority were set in high-income countries. Strategies analysed included cascade screening (23 strategies), opportunistic screening (13 strategies), systematic screening (11 strategies) and population-wide screening (15 strategies). Most of the strategies relied on genetic diagnosis for case ascertainment. The most common comparator was no screening, but some studies compared the proposed strategy versus current screening strategies or versus the best next alternative. Six studies evaluated screening in children while the remaining were targeted at adults. From a healthcare perspective, cascade screening was cost-effective in 78% of the studies [cost-adapted incremental cost-effectiveness ratios (ICERs) ranged from dominant to 2022 US$ 104,877], opportunistic screening in 85% (ICERs from US$4959 to US$41,705), systematic screening in 80% (ICERs from US$2763 to US$69,969) and population-wide screening in 60% (ICERs from US$1484 to US$223,240). The most common driver of ICER identified in the sensitivity analysis was the long-term cost of lipid-lowering treatment.
conclusionsBased on reported willingness to pay thresholds for each setting, most CEA studies concluded that screening for FH compared with no screening was cost-effective, regardless of the screening strategy. Cascade screening resulted in the largest health benefits per person tested.
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