Literature DB >> 23743435

Valuing the benefits of genetic testing for retinitis pigmentosa: a pilot application of the contingent valuation method.

Martin Eden1, Katherine Payne, Ryan M Combs, Georgina Hall, Marion McAllister, Graeme C M Black.   

Abstract

BACKGROUND: Technological advances present an opportunity for more people with, or at risk of, developing retinitis pigmentosa (RP) to be offered genetic testing. Valuation of these tests using current evaluative frameworks is problematic since benefits may be derived from diagnostic information rather than improvements in health. This pilot study aimed to explore if contingent valuation method (CVM) can be used to value the benefits of genetic testing for RP.
METHODS: CVM was used to elicit willingness-to-pay (WTP) values for (1) genetic counselling and (2) genetic counselling with genetic testing. Telephone and face-to-face interviews with a purposive sample of individuals with (n=25), and without (n=27), prior experience of RP were used to explore the feasibility and validity of CVM in this context.
RESULTS: Faced with a hypothetical scenario, the majority of participants stated that they would seek genetic counselling and testing in the context of RP. Between participant groups, respondents offered similar justifications for stated WTP values. Overall stated WTP was higher for genetic counselling plus testing (median=£524.00) compared with counselling alone (median=£224.50). Between-group differences in stated WTP were statistically significant; participants with prior knowledge of the condition were willing to pay more for genetic ophthalmology services.
CONCLUSIONS: Participants were able to attach a monetary value to the perceived potential benefit that genetic testing offered regardless of prior experience of the condition. This exploratory work represents an important step towards evaluating these services using formal cost-benefit analysis.

Entities:  

Keywords:  Diagnostic tests/Investigation; Genetics; Retina

Mesh:

Year:  2013        PMID: 23743435     DOI: 10.1136/bjophthalmol-2012-303020

Source DB:  PubMed          Journal:  Br J Ophthalmol        ISSN: 0007-1161            Impact factor:   4.638


  8 in total

1.  Variation in healthcare services for specialist genetic testing and implications for planning genetic services: the example of inherited retinal dystrophy in the English NHS.

Authors:  Mark Harrison; Stephen Birch; Martin Eden; Simon Ramsden; Tracey Farragher; Katherine Payne; Georgina Hall; Graeme Cm Black
Journal:  J Community Genet       Date:  2015-01-09

2.  Identifying variation in models of care for the genomic-based diagnosis of inherited retinal dystrophies in the United Kingdom.

Authors:  M Eden; K Payne; C Jones; S J Wright; G Hall; M McAllister; G Black
Journal:  Eye (Lond)       Date:  2016-04-15       Impact factor: 3.775

3.  The price of whole-genome sequencing may be decreasing, but who will be sequenced?

Authors:  Deborah A Marshall; Karen V MacDonald; Jill Oliver Robinson; Lisa F Barcellos; Milena Gianfrancesco; Monica Helm; Amy McGuire; Robert C Green; Michael P Douglas; Michael A Goldman; Kathryn A Phillips
Journal:  Per Med       Date:  2017-05-23       Impact factor: 2.512

Review 4.  Navigating the current landscape of clinical genetic testing for inherited retinal dystrophies.

Authors:  Kristy Lee; Seema Garg
Journal:  Genet Med       Date:  2015-03-19       Impact factor: 8.822

Review 5.  Conceptualising 'Benefits Beyond Health' in the Context of the Quality-Adjusted Life-Year: A Critical Interpretive Synthesis.

Authors:  Lidia Engel; Stirling Bryan; David G T Whitehurst
Journal:  Pharmacoeconomics       Date:  2021-08-23       Impact factor: 4.981

6.  Developing new frameworks to value genomic information: accounting for complexity.

Authors:  Martin Eden
Journal:  Per Med       Date:  2021-05-11       Impact factor: 2.512

7.  Validity and cost-effectiveness of cone adaptation test as a screening tool to detect retinitis pigmentosa.

Authors:  Rahul Deshpande; Prajakta Save; Madan Deshpande; Mahadev Shegunashi; Marium Chougule; Rajiv Khandekar
Journal:  Oman J Ophthalmol       Date:  2016 Sep-Dec

8.  Willingness to pay for genetic testing for inherited retinal disease.

Authors:  Sandy Tubeuf; Thomas A Willis; Barbara Potrata; Hilary Grant; Matthew J Allsop; Mushtaq Ahmed; Jenny Hewison; Martin McKibbin
Journal:  Eur J Hum Genet       Date:  2014-06-11       Impact factor: 4.246

  8 in total

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