Leonardi-Bee, Jo, Boateng, Christabel, Faria, Rita orcid.org/0000-0003-3410-1435 et al. (3 more authors) (2021) Effectiveness of cascade testing strategies in relatives for familial hypercholesterolemia:a systematic review and meta-analysis. Atherosclerosis. pp. 7-14. ISSN 0021-9150
Abstract
Background and aims: Cascade testing in relatives of index cases is the most cost-effective approach to identifying people with familial hypercholesterolemia (FH); however, it is currently unclear which strategy to contact relatives would be the most effective. A systematic review was performed to quantify the effectiveness of different strategies in cascade testing of FH. Methods: Comprehensive searches of three electronic databases and grey literature sources were searched (from inception to May 2020). Screening, data extraction and assessments of methodological quality were made independently by two reviewers. Meta-analyses of proportions were performed using random effects models. Effect measures are reported as percentages with 95% confidence intervals. Results: 24 non-comparative studies were included, of which 11 used a direct, 8 used indirect, and 5 used a combination of both direct and indirect cascade strategies. The median number of new relatives with FH per known index case was approximately 1. The combination strategy resulted in the largest yields of relatives tested for FH out of those contacted (40%, 95% CI 37% to 42%, 1 study) and relatives responding to testing out of those contacted (54%, 1 study); however, the direct strategy had the largest yield of index cases participating in cascade testing out of those with FH confirmed (94%, 8 studies) compared to other strategies (p0.01 for all comparisons). Conclusions: Evidence is limited; however, a combination strategy, which allows the index case to decide on method of contacting relatives, appears to lead to better yields compared to using the direct or indirect strategy.
Metadata
Item Type: | Article |
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Authors/Creators: |
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Copyright, Publisher and Additional Information: | © 2021 Published by Elsevier B.V. |
Dates: |
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Institution: | The University of York |
Academic Units: | The University of York > Faculty of Social Sciences (York) > Centre for Health Economics (York) The University of York > Faculty of Sciences (York) > Health Sciences (York) |
Depositing User: | Pure (York) |
Date Deposited: | 23 Sep 2021 08:50 |
Last Modified: | 16 Oct 2024 17:53 |
Published Version: | https://doi.org/10.1016/j.atherosclerosis.2021.09.... |
Status: | Published |
Refereed: | Yes |
Identification Number: | 10.1016/j.atherosclerosis.2021.09.014 |
Open Archives Initiative ID (OAI ID): | oai:eprints.whiterose.ac.uk:178454 |
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Filename: FH_cascade_paper_Leonardi_Bee_Atherosclerosis_accepted_version_with_attachments_.pdf
Description: FH cascade paper (Leonardi-Bee Atherosclerosis) (accepted version with attachments)
Licence: CC-BY-NC-ND 2.5