A cost-effectiveness evaluation of newborn hemoglobinopathy screening from the perspective of state health care systems

Gessner, Bradford D.; Teutsch, Steven M.; Shaffer, Phaedra A.

doi:10.1016/0378-3782(96)01761-6

Cited by 28 publications

(16 citation statements)

References 58 publications

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“…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%

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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: Existing Economic Evaluationsmentioning

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%

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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“…In particular, the costeffectiveness of universal screening for sickle cell disease in areas of low prevalence has been questioned on the basis of the low yield and high ICER associated with screening children from groups with a low frequency of sickle cell trait (37,126). All except one state currently screen all infants for hemoglobin disorders on the basis of evidence from a randomized trial that children with sickle cell anemia who received prophylactic antibiotic therapy had substantially lower risks of potentially lethal blood infections (36).…”

Section: Newborn Screeningmentioning

confidence: 99%

Lessons from Cost-Effectiveness Research for United States Public Health Policy

Grosse

Teutsch

Haddix

2007

Annu. Rev. Public Health

Self Cite

111

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The application of cost-effectiveness analysis to health care has been the subject of previous reviews. We address the use of economic evaluation methods in public health, including case studies of population-level policies, e.g., environmental regulations, injury prevention, tobacco control, folic acid fortification, and blood product safety, and the public health promotion of clinical preventive services, e.g., newborn screening, cancer screening, and childhood immunizations. We review the methods used in cost-effectiveness analysis, the implications for cost-effectiveness findings, and the extent to which economic studies have influenced policy and program decisions. We discuss reasons for the relatively limited impact to date of economic evaluation in public health. Finally, we address the vexing question of how to decide which interventions are cost effective and worthy of funding. Policy makers have funded certain interventions with rather high cost-effectiveness ratios, notably nucleic acid testing for blood product safety. Cost-effectiveness estimates are a decision aid, not a decision rule. 365

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“…Se diagnosticada precocemente, a criança poderá receber tratamento adequado nos primeiros anos de vida, reduzindo as complicações que decorrem da doença (3,18,19).…”

Section: Introductionunclassified

Investigação de hemoglobinopatias em sangue de cordão umbilical de recém-nascidos do Hospital de Base de São José do Rio Preto

Ducatti¹,

Teixeira²,

Galão³

et al. 2001

Rev. Bras. Hematol. Hemoter.

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A cost-effectiveness evaluation of newborn hemoglobinopathy screening from the perspective of state health care systems

Cited by 28 publications

References 58 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

Lessons from Cost-Effectiveness Research for United States Public Health Policy

Investigação de hemoglobinopatias em sangue de cordão umbilical de recém-nascidos do Hospital de Base de São José do Rio Preto

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