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Are whole-exome and whole-genome sequencing approaches cost-effective? A systematic review of the literature

Abstract:

Purpose: We conducted a systematic literature review to summarize the current health economic evidence for whole-exome sequencing (WES) and whole-genome sequencing (WGS).

Methods: Relevant studies were identified in the EMBASE, MEDLINE, Cochrane Library, EconLit and University of York Centre for Reviews and Dissemination databases from January 2005 to July 2016. Publications were included in the review if they were economic evaluations, cost studies, or outcome studies.

Results: Thirty-six studies met our inclusion criteria. These publications investigated the use of WES and WGS in a variety of genetic conditions in clinical practice, the most common being neurological or neurodevelopmental disorders. Study sample size varied from a single child to 2,000 patients. Cost estimates for a single test ranged from $555 to $5,169 for WES and from $1,906 to $24,810 for WGS. Few cost analyses presented data transparently and many publications did not state which components were included in cost estimates.

Conclusion: The current health economic evidence base to support the more widespread use of WES and WGS in clinical practice is very limited. Studies that carefully evaluate the costs, effectiveness, and cost-effectiveness of these tests are urgently needed to support their translation into clinical practice.

Publication status:
Published
Peer review status:
Peer reviewed

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Publisher copy:
10.1038/gim.2017.247

Authors


More by this author
Institution:
University of Oxford
Division:
Medical Sciences Division
Department:
Nuffield Department of Population Health; Population Health
Role:
Author
More by this author
Institution:
University of Oxford
Division:
Medical Sciences Division
Department:
Nuffield Department of Population Health; Population Health
Oxford college:
Oriel College
Role:
Author
ORCID:
0000-0003-2528-0638
More by this author
Institution:
University of Oxford
Division:
MSD
Department:
NDM
Sub department:
Human Genetics Wt Centre
Role:
Author
ORCID:
0000-0003-3602-5704
More by this author
Institution:
University of Oxford
Division:
Medical Sciences Division
Department:
Nuffield Department of Population Health; Population Health
Role:
Author


Publisher:
Nature Publishing Group
Journal:
Genetics in Medicine More from this journal
Volume:
20
Pages:
1122–1130
Publication date:
2018-02-15
Acceptance date:
2017-11-27
DOI:
EISSN:
1530-0366
ISSN:
1098-3600
Pmid:
29446766


Language:
English
Keywords:
Pubs id:
pubs:825633
UUID:
uuid:8cf329bc-73f9-41c0-b27b-3d944f8f9096
Local pid:
pubs:825633
Source identifiers:
825633
Deposit date:
2018-02-28

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