Evidence map›Paper›PMID 33925765›Full record

ArticleChildren (Basel, Switzerland)2021

Incorporating Cascade Effects of Genetic Testing in Economic Evaluation: A Scoping Review of Methodological Challenges.

Alexandra Cernat, Robin Z Hayeems, Lisa A Prosser, Wendy J Ungar

Abstract readScoping Review
In one paragraph

Article in Children (Basel, Switzerland), 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 11 papers.

0numbers the graph read from it
0cells of the map it votes in
11citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

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.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

11 citing papers in PubMed.

  1. Review
  2. Article
  3. Article
  4. Article
  5. Article
  6. Article
  7. Article
  8. Access to novel drugs and therapeutics for children and youth: Eliciting citizens' values to inform public funding decisions.Health expectations : an international journal of public participation in health care and health policy · 2023
    Article
  9. Article
  10. Article
  11. Assessing Value in Child Health.Children (Basel, Switzerland) · 2021
    Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

4 authors.

Alexandra CernatChild Health Evaluative Sciences, Hospital for Sick Children, Toronto, ON M5G 0A4, Canada.ORCID 0000-0001-6007-7825
Robin Z HayeemsChild Health Evaluative Sciences, Hospital for Sick Children, Toronto, ON M5G 0A4, Canada.ORCID 0000-0003-3269-8004
Lisa A ProsserSusan B. Meister Child Health Evaluation and Research Center, Department of Pediatrics, University of Michigan Medical School, Ann Arbor, MI 48109, USA.
Wendy J UngarChild Health Evaluative Sciences, Hospital for Sick Children, Toronto, ON M5G 0A4, Canada.ORCID 0000-0002-0762-0101

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Cascade genetic testing is indicated for family members of individuals testing positive on a genetic test, and is particularly relevant for child health because of their vulnerability and the long-term health and economic implications. Cascade testing has patient- and health system-level implications; however cascade costs and health effects are not routinely considered in economic evaluation. The methodological challenges associated with incorporating cascade effects in economic evaluation require examination. The purpose of this scoping review was to identify published economic evaluations that considered cascade genetic testing. Citation databases were searched for English-language economic evaluations reporting on cascade genetic testing. Nineteen publications were included. In four, genetic testing was used to identify new index patients-cascade effects were also considered; thirteen assessed cascade genetic testing strategies for the identification of at-risk relatives; and two calculated the costs of cascade genetic testing as a secondary objective. Methodological challenges associated with incorporating cascade effects in economic evaluation are related to study design, costing, measurement and valuation of health outcomes, and modeling. As health economic studies may currently be underestimating both the cost and health benefits attributable to genetic technologies through omission of cascade effects, development of methods to address these difficulties is required.

Indexed as

cascade testingeconomic evaluationgenetic testingmethodologyscoping review

Identifiers

PMID33925765
PMCPMC8145875

What OpenQuestion holds

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.