The Cost-Effectiveness Of Screening For Severe Combined Immunodeficiency (SCID) In The UK NHS Newborn Bloodspot Screening Programme

Severe combined immunodeficiency (SCID) can be detected through newborn bloodspot screening. In the UK, the National Screening Committee (NSC) requires screening programmes to be cost-effective at standard UK thresholds. To assess the cost-effectiveness of SCID screening for the NSC, a decision-tree model with lifetable estimates of outcomes was built. Model structure and parameterisation were informed by systematic review and expert clinical judgment. A public service perspective was used and lifetime costs and quality-adjusted life years (QALYs) were discounted at 3.5%. Probabilistic, one-way sensitivity analyses and an exploratory disbenefit analysis for the identification of non-SCID patients were conducted. Screening for SCID was estimated to result in an incremental cost-effectiveness ratio (ICER) of £18,222 with a reduction in SCID mortality from 8.1 (5–12) to 1.7 (0.6–4.0) cases per year of screening. Results were sensitive to a number of parameters, including the cost of the screening test, the incidence of SCID and the disbenefit to the healthy at birth and false-positive cases. Screening for SCID is likely to be cost-effective at £20,000 per QALY, key uncertainties relate to the impact on false positives and the impact on the identification of children with non-SCID T Cell lymphopenia.

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The Cost-Effectiveness of Expanding the UK Newborn Bloodspot Screening Programme to Include Five Additional Inborn Errors of Metabolism

International Journal of Neonatal Screening ◽

10.3390/ijns6040093 ◽

2020 ◽

Vol 6 (4) ◽

pp. 93

Author(s):

Alice Bessey ◽

James Chilcott ◽

Abdullah Pandor ◽

Suzy Paisley

Keyword(s):

Cost Effectiveness ◽

Social Services ◽

Inborn Errors Of Metabolism ◽

Screening Programme ◽

Tree Model ◽

Inborn Errors ◽

Life Years ◽

Newborn Bloodspot Screening ◽

The Uk ◽

The Cost

Glutaric aciduria type 1, homocystinuria, isovaleric acidaemia, long-chain hydroxyacyl CoA dehydrogenase deficiency and maple syrup urine disease are all inborn errors of metabolism that can be detected through newborn bloodspot screening. This evaluation was undertaken in 2013 to provide evidence to the UK National Screening Committee for the cost-effectiveness of including these five conditions in the UK Newborn Bloodspot Screening Programme. A decision-tree model with lifetable estimates of outcomes was built with the model structure and parameterisation informed by a systematic review and expert clinical judgment. A National Health Service/Personal Social Services perspective was used, and lifetime costs and quality-adjusted life years (QALYs) were discounted at 1.5%. Uncertainty in the results was explored using expected value of perfect information analysis methods together with a sensitivity analysis using the screened incidence rate in the UK from 2014 to 2018. The model estimates that screening for all the conditions is more effective and cost saving when compared to not screening for each of the conditions, and the results were robust to the updated incidence rates. The key uncertainties included the sensitivity and specificity of the screening test and the estimated costs and QALYs.

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Modelling the cost-effectiveness and capacity impact of changes to colposcopy referral guidelines for women with mild dyskaryosis in the UK Cervical Screening Programme

BJOG An International Journal of Obstetrics & Gynaecology ◽

10.1111/j.1471-0528.2008.01683.x ◽

2008 ◽

Vol 115 (6) ◽

pp. 749-757 ◽

Cited By ~ 3

Author(s):

R Hadwin ◽

S Eggington ◽

A Brennan ◽

P Walker ◽

J Patnick ◽

...

Keyword(s):

Cost Effectiveness ◽

Screening Programme ◽

Cervical Screening ◽

The Uk ◽

The Cost

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Evaluating the Cost-Effectiveness of Changes to the Surveillance Intervals in the UK Abdominal Aortic Aneurysm Screening Programme

Value in Health ◽

10.1016/j.jval.2020.10.015 ◽

2020 ◽

Author(s):

Michael J. Sweeting ◽

John Marshall ◽

Matthew Glover ◽

Akhtar Nasim ◽

Matthew J. Bown

Keyword(s):

Abdominal Aortic Aneurysm ◽

Cost Effectiveness ◽

Aortic Aneurysm ◽

Screening Programme ◽

Abdominal Aortic ◽

The Uk ◽

The Cost

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Economic Evaluation of Different Screening Strategies for Severe Combined Immunodeficiency Based on Real-Life Data

International Journal of Neonatal Screening ◽

10.3390/ijns7030060 ◽

2021 ◽

Vol 7 (3) ◽

pp. 60

Author(s):

M. Elske van den Akker-van Marle ◽

Maartje Blom ◽

Mirjam van der Burg ◽

Robbert G. M. Bredius ◽

Catharina P. B. Van der Ploeg

Keyword(s):

Cost Effectiveness ◽

Severe Combined Immunodeficiency ◽

Real Life ◽

Final Diagnosis ◽

Screening Strategy ◽

Combined Immunodeficiency ◽

Life Data ◽

Real Life Data ◽

Screening Strategies ◽

The Cost

Although several countries have adopted severe combined immunodeficiency (SCID) into their newborn screening (NBS) program, other countries are still in the decision process of adding this disorder in their program and finding the appropriate screening strategy. This decision may be influenced by the cost(-effectiveness) of these screening strategies. In this study, the cost(-effectiveness) of different NBS strategies for SCID was estimated based on real-life data from a prospective implementation study in the Netherlands. The cost of testing per child for SCID was estimated at EUR 6.36. The cost of diagnostics after screen-positive results was assessed to vary between EUR 985 and 8561 per child dependent on final diagnosis. Cost-effectiveness ratios varied from EUR 41,300 per QALY for the screening strategy with T-cell receptor excision circle (TREC) ≤ 6 copies/punch to EUR 44,100 for the screening strategy with a cut-off value of TREC ≤ 10 copies/punch. The analysis based on real-life data resulted in higher costs, and consequently in less favorable cost-effectiveness estimates than analyses based on hypothetical data, indicating the need for verifying model assumptions with real-life data. The comparison of different screening strategies suggest that strategies with a lower number of referrals, e.g., by distinguishing between urgent and less urgent referrals, are favorable from an economic perspective.

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Study Design for an Evaluation of Newborn Screening for SCID in the UK

International Journal of Neonatal Screening ◽

10.3390/ijns8010004 ◽

2022 ◽

Vol 8 (1) ◽

pp. 4

Author(s):

David Elliman

Keyword(s):

False Positive ◽

Screening Programme ◽

Severe Combined Immunodeficiency ◽

Cost Effective ◽

Cell Receptor ◽

Premature Babies ◽

Separate Pathway ◽

Excision Circles ◽

Newborn Bloodspot Screening ◽

The Uk

Severe combined immunodeficiency is a rare inherited disorder, which, if untreated, invariably proves fatal in late infancy or early childhood. With treatment, the prognosis is much improved. Early treatment of the siblings of cases, before they become symptomatic, has shown considerable improvements in outcomes. Based on this and the development of a test that can be used on the whole population of neonates (measurement of T-cell receptor excision circles—TRECs), many countries have added it to their routine newborn bloodspot screening programmes. The UK National Screening Committee (UKNSC) has considered whether SCID should be added to the UK screening programme and concluded that it was likely to be cost effective, but that there were a number of uncertainties that should be resolved before a national roll-out could be recommended. These include some aspects of the test, such as: cost; the use of different assays and cut-off levels to reduce false positive rates, while maintaining sensitivity; the overall benefits of screening for disease outcome in patients with SCID and other identified disorders; the need for a separate pathway for premature babies; the acceptability of the screening programme to parents of babies who have normal and abnormal (both true and false positive) screening results. To achieve this, screening of two thirds of babies born in England over a two-year period has been planned, beginning in September 2021. The outcomes and costs of care of babies identified by the screening will be compared with those of babies identified with SCID in the rest of the UK. The effect of the screening programme on parents will form part of a separate research project.

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Screening for Melanoma by Primary Health Care Physicians: A Cost—Effectiveness Analysis

Journal of Medical Screening ◽

10.1177/096914139600300112 ◽

1996 ◽

Vol 3 (1) ◽

pp. 47-53 ◽

Cited By ~ 50

Author(s):

Afaf Girgis ◽

Philip Clarke ◽

Robert C Burton ◽

Rob W Sanson—Fisher

Keyword(s):

Cost Effectiveness ◽

Screening Programme ◽

Cost Effective ◽

Economic Assessment ◽

Screening Tests ◽

Family Practitioners ◽

Effectiveness Analysis ◽

The Cost ◽

Primary Health Care Physicians ◽

Melanoma Screening

Background and design— Australia has the highest rates of skin cancer in the world, and the incidence is estimated to be doubling every 10 years. Despite advances in the early detection and treatment of melanoma about 800 people still die nationally of the disease each year. A possible strategy for further reducing the mortality from melanoma is an organised programme of population screening for unsuspected lesions in asymptomatic people. Arguments against introducing melanoma screening have been based on cost and the lack of reliable data on the efficacy of any screening tests. To date, however, there has been no systematic economic assessment of the cost effectiveness of melanoma screening. The purpose of this research was to determine whether screening may be potentially cost effective and, therefore, warrants further investigation. A computer was used to simulate the effects of a hypothetical melanoma screening programme that was in operation for 20 years, using cohorts of Australians aged 50 at the start of the programme. Based on this simulation, cost—effectiveness estimates of melanoma screening were calculated. Results— Under the standard assumptions used in the model, and setting the sensitivity of the screening test (visual inspection of the skin) at 60%, cost effectiveness ranged from Aust$6853 per life year saved for men if screening was undertaken five yearly to $12137 if screening was two yearly. For women, it ranged from $11 102 for five yearly screening to $20 877 for two yearly screening. Conclusion— The analysis suggests that a melanoma screening programme could be cost effective, particularly if five yearly screening is implemented by family practitioners for men over the age of 50.

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