Costs, effects, and savings of screening for cystic fibrosis gene carriers

Wildhagen, M. F.; Hilderink, Henk; Verzijl, J. G.; Verheij, J. B. G. M.; Kooij, L; Tijmstra, T; Kate, Leo P. ten; Habbema, J. Dik F.

doi:10.1136/jech.52.7.459

Cited by 35 publications

(41 citation statements)

References 49 publications

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“…A systematic review of published economic evaluations of NBS (reported in Chapter 6) identified five studies that included a cost associated with information provision related to NBS. [119][120][121][122][123] In the two identified studies relevant to the UK setting, 119,120 an incremental cost of providing information, based on the additional time that it would take a midwife to explain the additional tests being evaluated, was used (£0.30 119 and £0.40 120 ). Pandor et al 119 provided little information on how the cost they used was derived, but did refer to being informed by a consultation with a midwife.…”

Section: Discussionmentioning

confidence: 99%

“…In a study carried out in Australia, Geelhoed et al 121 used a similar method, multiplying the salary cost for a midwife by the amount of time spent providing information. Wildhagen et al 122 estimated the cost of providing information about neonatal CF carrier screening by taking a value from a previous study 124 and scaling it by a factor of 60%. This study used a top-down approach that provided little information about how individual items of resource use were valued.…”

Section: Discussionmentioning

confidence: 99%

“…Five of the studies compared the NBSP with no screening. 122,[163][164][165][166] The remaining seven studies compared screening with existing strategies. Seven studies discussed the implications of using screening strategies such as universal or targeted strategies.…”

Section: Existing Economic Evaluationsmentioning

confidence: 99%

“…Seven studies discussed the implications of using screening strategies such as universal or targeted strategies. 121,[163][164][165][168][169][170] Three studies focused on expanded screening programmes, including additional screening for MCADD, 42,139,167 and two studies reported screening strategies specifically for CF 122 and MCADD. 166 Around half of the identified studies focused on understanding the economic impact of newborn screening as a national programme.…”

Section: Existing Economic Evaluationsmentioning

confidence: 99%

“…An individual figure for the cost of providing information was not given but it can be calculated by dividing the total cost of nursing input in the sample (AU$326,875) by the number of babies seen (25,000) and multiplying this by the proportion of nurse time spent giving information (0.5), resulting in a value of AU$6.54 (2001 prices) per child screened. Wildhagen et al, 122 based in the Netherlands, evaluated a CF NBSP but calculated the cost of information provision by using the mass media costs of information provision for a breast cancer screening programme. 124 The analysis assumed that the relative cost of providing a NBSP compared with a breast cancer screening programme would be reduced by 40% to account for the greater ease of introducing NBS into clinical practice.…”

Section: Existing Evidence On the Costs Of Information Provisionmentioning

confidence: 99%

See 4 more Smart Citations

Provision of information about newborn screening antenatally: a sequential exploratory mixed-methods project

Ulph

Wright

Dharni

et al. 2017

Health Technol Assess

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BackgroundParticipation in the UK Newborn Bloodspot Screening Programme (NBSP) requires parental consent but concerns exist about whether or not this happens in practice and the best methods and timing to obtain consent at reasonable cost.ObjectivesTo collate all possible modes of prescreening communication and consent for newborn (neonatal) screening (NBS); examine midwives’, screening professionals’ and users’ views about the feasibility, efficiency and impact on understanding of each; measure midwives’ and parents’ preferences for information provision; and identify key drivers of cost-effectiveness for alternative modes of information provision.DesignSix study designs were used: (1) realist review – to generate alternative communication and consent models; (2) qualitative interviews with parents and health professionals – to examine the implications of current practice for understanding and views on alternative models; (3) survey and observation of midwives – to establish current costs; (4) stated preference surveys with midwives, parents and potential future parents – to establish preferences for information provision; (5) economic analysis – to identify cost-effectiveness drivers of alternative models; and (6) stakeholder validation focus groups and interviews – to examine the acceptability, views and broader impact of alternative communication and consent models.SettingProviders and users of NBS in England.ParticipantsStudy 2: 45 parents and 37 health professionals; study 3: 22 midwives and eight observations; study 4: 705 adults aged 18–45 years and 134 midwives; and study 6: 12 health-care professionals and five parents.ResultsThe realist review identified low parental knowledge and evidence of coercive consent practices. Interview, focus group and stated preference data suggested a preference for full information, with some valuing this more than choice. Health professionals preferred informed choice models but parents and health professionals queried whether or not current consent was fully informed. Barriers to using leaflets effectively were highlighted. All studies indicated that a ‘personalised’ approach to NBS communication, allowing parents to select the mode and level of information suited to their learning needs, could have added value. A personalised approach should rely on midwife communication and should occur in the third trimester. Overall awareness was identified as requiring improvement. Starting NBS communication by alerting parents that they have a choice to make and telling them that samples could be stored are both likely to enhance engagement. The methods of information provision and maternal anxiety causing additional visits to health-care professionals were the drivers of relative cost-effectiveness. Lack of data to populate an economic analysis, confirmed by value of information analysis, indicated a need for further research.LimitationsThere are some limitations with regard to the range of participants used in studies 2 and 3 and so caution should be exercised when interpreting some of the results.ConclusionsThis project highlighted the importance of focusing on information receipt and identified key communication barriers. Health professionals strongly preferred informed consent, which parents endorsed if they were made aware of sample storage. Uniform models of information provision were perceived as ineffective. A choice of information provision was supported by health professionals and parents, which both enhances cost-effectiveness and improves engagement, understanding and the validity of consent. Remaining uncertainties suggest that more research is needed before new communication modes are introduced into practice. Future research should measure the impact of the suggested practice changes (informing in third trimester, information toolkits, changed role of midwife).Trial registrationCurrent Controlled Trials ISRCTN70227207.FundingThis project was funded by the NIHR Health Technology Assessment programme and will be published in full inHealth Technology Assessment; Vol. 21, No. 55. See the NIHR Journals Library website for further project information.

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Existing Economic Evaluationsmentioning

confidence: 99%

Section: Existing Economic Evaluationsmentioning

confidence: 99%

Section: Existing Evidence On the Costs Of Information Provisionmentioning

confidence: 99%

See 3 more Smart Citations

Provision of information about newborn screening antenatally: a sequential exploratory mixed-methods project

Ulph

Wright

Dharni

et al. 2017

Health Technol Assess

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Preconception care: Challenge of the new millennium?

Schrander‐Stumpel

1999

Am. J. Med. Genet.

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Couples who plan their pregnancies can elect anticipatory care with the aim of improved health for mother and child. While it has proved to be beneficial in such specific disorders as maternal diabetes mellitus, preconception care is not yet established as a part of primary care. Preconception care is a form of community genetics and consists of three main components: risk assessment, health promotion, and intervention. Intake of folic acid during the time surrounding conception is important in preventing neural tube defects and other congenital anomalies. Primary care health professionals may be involved in providing preconception care, as may be gynecologists, midwives, nurses, clinical geneticists, and genetic counselors. Several questions arose from a Dutch pilot study. Is there a need for preconception care? Do women want this care? Can positive effects (and cost-effectiveness) be documented? How best are the parents-to-be contacted? Last, but not least, who should provide the care?

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Genetic testing in the European Union: does economic evaluation matter?

et al. 2011

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Economic evaluation of genetic technologies matters but the number of published studies is still rather low as to be widely used for most of the decisions in different jurisdictions across the EU. Further, the decision bodies across EU27 are fragmented and the responsibilities are located at different levels of the decision process for what it is difficult to find out whether a given decision on genetic tests was somehow supported by the economic evaluation results.

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Costs, effects, and savings of screening for cystic fibrosis gene carriers

Abstract: Abstract

Cited by 35 publications

References 49 publications

Provision of information about newborn screening antenatally: a sequential exploratory mixed-methods project

Provision of information about newborn screening antenatally: a sequential exploratory mixed-methods project

Preconception care: Challenge of the new millennium?

Genetic testing in the European Union: does economic evaluation matter?

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