The Cost-Effectiveness of Expanding Newborn Screening for up to 21 Inherited Metabolic Disorders Using Tandem Mass Spectrometry: Results from a Decision-Analytic Model

Cipriano, Lauren E.; Rupar, C. Anthony; Zaric, Gregory S.

doi:10.1111/j.1524-4733.2006.00156.x

Cited by 65 publications

(60 citation statements)

References 71 publications

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“…Two subsequent studies also used the disutility value of 0.03 used by Venditti et al 137 In an economic analysis of an expanded NBSP, the authors allowed the effect of parental anxiety to vary from a disutility of 0.01 to a value of 0.05 (a QALY loss of 0.0125). 138 This study did not report the impact of including this disutility on the relative cost-effectiveness of the expanded NBSP. In a study by Cipriano et al, 138 based in Canada, including a disutility for parental anxiety appeared to have a significant impact on the relative cost-effectiveness of expanding a NBSP.…”

Section: Existing Evidence On the Harms Of Information Provisionmentioning

confidence: 99%

“…In a study by Cipriano et al, 138 based in Canada, including a disutility for parental anxiety appeared to have a significant impact on the relative cost-effectiveness of expanding a NBSP. 138 Including the disutility value caused the intervention to not be cost-effectiveness when 15 conditions were included in the NBSP.…”

Section: Existing Evidence On the Harms Of Information Provisionmentioning

confidence: 99%

“…Four studies were identified in the systematic review that included a utility decrement for parents whose child had received a false-positive result. 42,[137][138][139] These disutility values ranged between 0.01 and 0.05. In this model, the mean of the reported published values (0.018) was used for the utility decrement in the base-case analysis.…”

Section: Consequencesmentioning

confidence: 99%

See 2 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: Existing Evidence On the Harms Of Information Provisionmentioning

confidence: 99%

Section: Existing Evidence On the Harms Of Information Provisionmentioning

confidence: 99%

Section: Consequencesmentioning

confidence: 99%

See 1 more Smart Citation

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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“…[218,219] Also, there is no increased chance of recovery through urine screening in newborns for the presence of a neuroblastoma (malign neoformation of sympathetic nerve tissue), [220,221] whereas newborn screening of blood from a heel stick for possible metabolic disease (for example, phenylketonuria, hypothyroidism) is of definite benefit to the patient. [222,223] A subject of current discussion is expansion of the number of newborn screenings to 21 metabolic diseases with the aid of tandem MS. [224] A further step, the "lab-on-a-chip", offers the possibility of real-time observation of a vast array of blood parameters or cellular processes (optimistic authors speak in terms of thousands). [225][226][227] The large number of parameters, rapid availability, and simple acquisition of results leads to new problems, which are especially important for the clinical physician.…”

Section: Discussionmentioning

confidence: 99%

Clinical Chemistry: Challenges for Analytical Chemistry and the Nanosciences from Medicine

Durner

2010

Angew Chem Int Ed

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Clinical chemistry and laboratory medicine can look back over more than 150 years of eventful history. The subject encompasses all the medicinal disciplines as well as the remaining natural sciences. Clinical chemistry demonstrates how new insights from basic research in biochemical, biological, analytical chemical, engineering, and information technology can be transferred into the daily routine of medicine to improve diagnosis, therapeutic monitoring, and prevention. This Review begins with a presentation of the development of clinical chemistry. Individual steps between the drawing of blood and interpretation of laboratory data are then illustrated; here not only are pitfalls described, but so are quality control systems. The introduction of new methods and trends into medicinal analysis is explored, along with opportunities and problems associated with personalized medicine.

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“…B. ein rasches und kosteneffizientes Screening aller Neugeborenen auf mehr als 20 verschiedene angeborene Stoffwechselerkrankungen. Eine frühzeitige Erkennung und rechtzeitige Behandlung dieser Erkrankungen reduzieren dabei nicht nur deren Morbidität und Mortalität, sondern auch die damit verbundenen sozialen Belastungen [9].…”

Section: Introductionunclassified

Genetik in der Pädiatrie als Interaktion zwischen Klinik und Labor

Haas

Bodamer

2008

Monatsschr Kinderheilkd

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The Cost-Effectiveness of Expanding Newborn Screening for up to 21 Inherited Metabolic Disorders Using Tandem Mass Spectrometry: Results from a Decision-Analytic Model

Cited by 65 publications

References 71 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

Clinical Chemistry: Challenges for Analytical Chemistry and the Nanosciences from Medicine

Genetik in der Pädiatrie als Interaktion zwischen Klinik und Labor

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