Benefits and harms of antenatal and newborn screening programmes in health economic assessments: the VALENTIA systematic review and qualitative investigation.

Background: Health economic assessments are used to determine whether the resources needed to generate net benefit from an antenatal or newborn screening programme, driven by multiple benefits and harms, are justifiable. It is not known what benefits and harms have been adopted by economic evaluations assessing these programmes and whether they omit benefits and harms considered important to relevant stakeholders. Objectives: (1) To identify the benefits and harms adopted by health economic assessments in this area, and to assess how they have been measured and valued; (2) to identify attributes or relevance to stakeholders that ought to be considered in future economic assessments; and (3) to make recommendations about the benefits and harms that should be considered by these studies. Design: Mixed methods combining systematic review and qualitative work. Systematic review methods: We searched the published and grey literature from January 2000 to January 2021 using all major electronic databases. Economic evaluations of an antenatal or newborn screening programme in one or more Organisation for Economic Co-operation and Development countries were considered eligible. Reporting quality was assessed using the Consolidated Health Economic Evaluation Reporting Standards checklist. We identified benefits and harms using an integrative descriptive analysis and constructed a thematic framework. Qualitative methods: We conducted a meta-ethnography of the existing literature on newborn screening experiences, a secondary analysis of existing individual interviews related to antenatal or newborn screening or living with screened-for conditions, and a thematic analysis of primary data collected with stakeholders about their experiences with screening. Results: The literature searches identified 52,244 articles and reports, and 336 unique studies were included. Thematic framework resulted in seven themes: (1) diagnosis of screened for condition, (2) life-years and health status adjustments, (3) treatment, (4) long-term costs, (5) overdiagnosis, (6) pregnancy loss and (7) spillover effects on family members. Diagnosis of screened-for condition (115, 47.5%), life-years and health status adjustments (90, 37.2%) and treatment (88, 36.4%) accounted for most of the benefits and harms evaluating antenatal screening. The same themes accounted for most of the benefits and harms included in studies assessing newborn screening. Long-term costs, overdiagnosis and spillover effects tended to be ignored. The wide-reaching family implications of screening were considered important to stakeholders. We observed good overlap between the thematic framework and the qualitative evidence. Limitations: Dual data extraction within the systematic literature review was not feasible due to the large number of studies included. It was difficult to recruit healthcare professionals in the stakeholder's interviews. Conclusions: There is no consistency in the selection of benefits and harms used in health economic assessments in this area, suggesting that additional methods guidance is needed. Our proposed thematic framework can be used to guide the development of future health economic assessments evaluating antenatal and newborn screening programmes. Study registration: This study is registered as PROSPERO CRD42020165236. Funding: This award was funded by the National Institute for Health and Care Research (NIHR) Health Technology Assessment programme (NIHR award ref: NIHR127489) and is published in full in Health Technology Assessment; Vol. 28, No. 25. See the NIHR Funding and Awards website for further award information.

Every year the NHS offers pregnant women screening tests to assess the chances of them or their unborn baby having or developing a health condition. It also offers screening tests for newborn babies to look for a range of health conditions. The implementation of screening programmes and the care for women and babies require many resources and funding for the NHS, so it is important that screening programmes represent good value for money. This means that the amount of money the NHS spends on a programme is justified by the amount of benefit that the programme gives. We wanted to see whether researchers consider all the important benefits and harms associated with screening of pregnant women and newborn babies when calculating value for money. To do this, we searched all studies available in developed countries to identify what benefits and harms they considered. We also considered the views of parents and healthcare professionals on the benefits and harms screening that creates for families and wider society. We found that the identification of benefits and harms of screening is complex because screening results affect a range of people (mother­baby, parents, extended family and wider society). Researchers calculating the value for money of screening programmes have, to date, concentrated on a narrow range of benefits and harms and ignored many factors that are important to people affected by screening results. From our discussions with parents and healthcare professionals, we found that wider impacts on families are an important consideration. Only one study we looked at considered wider impacts on families. Our work also found that parent's ability to recognise, absorb and apply new information to understand their child's screening results or condition is important. Healthcare professionals involve in screening should consider this when supporting families of children with a condition. We have created a list for researchers to identify the benefits and harms that are important to include in future studies. We have also identified different ways researchers can value these benefits and harms, so they are incorporated into their studies in a meaningful way.

Neonatal health care costs of very preterm babies in England: a retrospective analysis of a national birth cohort.

OBJECTIVES: Babies born between 27+0 and 31+6 weeks of gestation represent the largest group of very preterm babies requiring National Health Service (NHS) care; however, up-to-date, cost figures for the UK are not currently available. This study estimates neonatal costs to hospital discharge for this group of very preterm babies in England. DESIGN: Retrospective analysis of resource use data recorded within the National Neonatal Research Database. SETTING: Neonatal units in England. PATIENTS: Babies born between 27+0 and 31+6 weeks of gestation in England and discharged from a neonatal unit between 2014 and 2018. MAIN OUTCOME MEASURES: Days receiving different levels of neonatal care were costed, along with other specialised clinical activities. Mean resource use and costs per baby are presented by gestational age at birth, along with total costs for the cohort. RESULTS: Based on data for 28 154 very preterm babies, the annual total costs of neonatal care were estimated to be £262 million, with 96% of costs attributable to routine daily care provided by units. The mean (SD) total cost per baby of this routine care varied by gestational age at birth; £75 594 (£34 874) at 27 weeks as compared with £27 401 (£14 947) at 31 weeks. CONCLUSIONS: Neonatal healthcare costs for very preterm babies vary substantially by gestational age at birth. The findings presented here are a useful resource to stakeholders including NHS managers, clinicians, researchers and policymakers.

Benefits and harms adopted by health economic assessments evaluating antenatal and newborn screening programmes in OECD countries: A systematic review of 336 articles and reports.

BACKGROUND: Health economic assessments are used to determine whether the resources needed to generate net benefit from a screening programme, driven by multiple complex benefits and harms, are justifiable. We systematically identified the benefits and harms incorporated within economic assessments evaluating antenatal and newborn screening programmes. METHODS: For this systematic review and thematic analysis, we searched the published and grey literature from January 2000 to January 2021. Studies that included an economic evaluation of an antenatal or newborn screening programme in an OECD country were eligible. We identified benefits and harms using an integrative descriptive analysis, and illustrated a thematic framework. (Systematic review registration PROSPERO, CRD42020165236). FINDINGS: The searches identified 52,244 articles and reports and 336 (242 antenatal and 95 newborn) were included. Eighty-six subthemes grouped into seven themes were identified: 1) diagnosis of screened for condition, 2) life years and health status adjustments, 3) treatment, 4) long-term costs, 5) overdiagnosis, 6) pregnancy loss, and 7) spillover effects on family members. Diagnosis of screened for condition (115 studies, 47.5%), life-years and health status adjustments (90 studies, 37.2%) and treatment (88 studies, 36.4%) accounted for most of the benefits and harms evaluating antenatal screening. The same themes accounted for most of the benefits and harms included in studies assessing newborn screening. Overdiagnosis and spillover effects tended to be ignored. INTERPRETATION: Our proposed framework can be used to guide the development of future health economic assessments evaluating antenatal and newborn screening programmes, to prevent exclusion of important potential benefits and harms.

Methods for evaluating the benefits and harms of antenatal and newborn screening programmes adopted by health economic assessments: protocol for a systematic review.

INTRODUCTION: Complex organisational arrangements are required to deliver antenatal and newborn screening programmes. Decision-makers consider the benefits and harms of screening when reviewing the evidence about these programmes. Economic evaluations contribute one important part of this assessment process. However, it is not fully understood what approaches health economic assessments have adopted to measure and value benefits and harms. This study aims to systematically review and critique the published and grey literature on methods for identifying, measuring and valuing the benefits and harms of antenatal and newborn screening adopted by health economic assessments. METHODS AND ANALYSIS: Nine bibliographic databases will be searched from 2000 onwards. These search strategies will be supplemented by manual reference searching of bibliographies, forward citation searching, contacts with experts, author searching and web searching for grey literature. Studies will be selected for review if they report health economic assessments of an antenatal or newborn screening programme. Assessments of title and abstracts and full reports will be undertaken independently with disagreements resolved through discussion. Data extraction will include fields to assess the reporting quality of the studies using the Consolidated Health Economic Evaluation Reporting Standards statement and a bespoke ancillary form to assess how benefits and harms have been accounted for. ETHICS AND DISSEMINATION: This is an evidence synthesis review from already published materials and hence ethics committee approval or written informed consent will not be required. Our results will be disseminated by publishing in high-impact peer-review journals and presenting at relevant conferences. PROSPERO REGISTRATION NUMBER: CRD42020165236.

Economic evaluation of patient direct access to NHS physiotherapy services.

OBJECTIVES: Our aim was to undertake an economic evaluation of patient direct access to physiotherapy in the UK NHS by comparing the number of patients treated, waiting time, cost and health gain from a direct access pathway versus traditional GP-referral to NHS physiotherapy. DESIGN: The authors used a discrete event simulation (DES) model to represent a hypothetical GP practice of 10,000 patients. Costs were measured from the perspective of the NHS and society. Outcomes were predicted waiting times, the total number of patients with musculoskeletal conditions who received physiotherapy and quality adjusted life years (QALYs) gained, each estimated over a one year period. Model inputs were based on a pilot cluster randomised controlled trial (RCT) conducted in four general practices in Cheshire, UK, and other sources from the literature. RESULTS: Direct access could increase the number of patients receiving at least one physiotherapy appointment by 63%, but without investment in extra physiotherapist capacity would increase waiting time dramatically. The increase in activity is associated with a cost of £4999 per QALY gained. CONCLUSIONS: Direct access to physiotherapy services would be cost-effective and benefit patients given current cost per QALY thresholds used in England. This is because physiotherapy itself is cost-effective, rather than through savings in GP time. Direct access without an increase in supply of physiotherapists would increase waiting times and would be unlikely to be cost saving for the NHS owing to the likely increase in the use of physiotherapy services.

The impact of social health insurance on rural populations.

Improving health outcomes of rural populations in low- and middle-income countries represents a significant challenge. A key part of this is ensuring access to health services and protecting households from financial risk caused by unaffordable medical care. In 2003, China introduced a heavily subsidised voluntary social health insurance programme that aimed to provide 800 million rural residents with access to health services and curb medical impoverishment. This paper provides new evidence on the impact of the scheme on health care utilisation and medical expenditure. Given the voluntary nature of the insurance enrolment, we exploit the uneven roll-out of the programme across rural counties as a natural experiment to explore causal inference. We find little effect of the insurance on the use of formal medical care and out-of-pocket health payments. However, there is evidence that it directed people away from informal health care towards village clinics, especially among patients with lower income. The insurance has also led to a reduction in the use of city hospitals among the rich. The shift to village clinics from informal care and higher-level hospitals suggests that the NRCMS has the potential to improve efficiency within the health care system and help patients to obtain less costly primary care. However, the poor quality of primary care and insufficient insurance coverage for outpatient services remains a concern.

Economic evaluation of computerised interpretation of fetal heart rate during labour: a cost-consequence analysis alongside the INFANT study.

OBJECTIVE: Economic evaluation of computerised decision-support software intended to assist in the interpretation of a cardiotocography (CTG) during birth. DESIGN: Individual patient level data from the INFANT study (an unmasked randomised controlled trial). SETTING: Maternity units in the UK and Ireland. POPULATION: Singleton or twin pregnancy women of 35 weeks' gestation or more and receiving continuous electronic fetal monitoring during labour. INTERVENTION: Computerised decision-support software. METHODS: Cost-consequence analysis presenting costs and outcomes with a time horizon of 2 years from a government healthcare perspective. Unit cost data collected from a combination of primary and secondary sources. MAIN OUTCOME MEASURES: Primary clinical outcomes were (i) composite 'poor neonatal outcome' and (ii) developmental assessment at age 2 years in a subset of surviving children. Mean cost per mother and infant dyad from birth to hospital discharge, and from hospital discharge to 24 months follow-up. Maternal health-related quality of life was assessed at 12 and 24 months follow-up using the EuroQol three-level health-related quality of life instrument (EQ-5D-3L). RESULTS: Data were analysed for 46 042 women and 46 614 infants. No statistically significant differences were detected between trial arms in any of the primary clinical outcomes or maternal quality of life. No statistically significant differences in costs were detected in maternal or infant costs from trial entry to hospital discharge or overall from hospital discharge to 2-year follow-up. CONCLUSIONS: Decision-support software during labour is not associated with additional maternal or infant benefits and over a 2-year period the software did not lead to additional costs or savings to the National Health Service. TRIAL REGISTRATION NUMBER: ISRCTN98680152.

Optimising neonatal service provision for preterm babies born between 27 and 31 weeks gestation in England (OPTI-PREM), using national data, qualitative research and economic analysis: a study protocol.

INTRODUCTION: In England, for babies born at 23-26 weeks gestation, care in a neonatal intensive care unit (NICU) as opposed to a local neonatal unit (LNU) improves survival to discharge. This evidence is shaping neonatal health services. In contrast, there is no evidence to guide location of care for the next most vulnerable group (born at 27-31 weeks gestation) whose care is currently spread between 45 NICU and 84 LNU in England. This group represents 12% of preterm births in England and over onr-third of all neonatal unit care days. Compared with those born at 23-26 weeks gestation, they account for four times more admissions and twice as many National Health Service bed days/year. METHODS: In this mixed-methods study, our primary objective is to assess, for babies born at 27-31 weeks gestation and admitted to a neonatal unit in England, whether care in an NICU vs an LNU impacts on survival and key morbidities (up to age 1 year), at each gestational age in weeks. Routinely recorded data extracted from real-time, point-of-care patient management systems held in the National Neonatal Research Database, Hospital Episode Statistics and Office for National Statistics, for January 2014 to December 2018, will be analysed. Secondary objectives are to assess (1) whether differences in care provided, rather than a focus on LNU/NICU designation, drives gestation-specific outcomes, (2) where care is most cost-effective and (3) what parents' and clinicians' perspectives are on place of care, and how these could guide clinical decision-making. Our findings will be used to develop recommendations, in collaboration with national bodies, to inform clinical practice, commissioning and policy-making. The project is supported by a parent advisory panel and a study steering committee. ETHICS AND DISSEMINATION: Research ethics approval has been obtained (IRAS 212304). Dissemination will be through publication of findings and development of recommendations for care. TRIAL REGISTRATION NUMBER: NCT02994849 and ISRCTN74230187.

Impact of the Southwark and Lambeth Integrated Care Older People's Programme on hospital utilisation and costs: controlled time series and cost-consequence analysis.

OBJECTIVES: To estimate the impact on hospital utilisation and costs of a multi-faceted primary care intervention for older people identified as being at risk of avoidable hospitalisation. DESIGN: Observational study: controlled time series analysis and estimation of costs and cost consequences of the Programme. General practitioner (GP)'s practice level data were analysed from 2009 to 2016 (intervention operated from 2012 to 2016). Mixed-effect Poisson regression models of hospital utilisation included comparisons with control practices and background trends in addition to within-practice comparisons. Cost estimation used standard tariff values. SETTING: 94 practices in Southwark and Lambeth and 263 control practices from other parts of England. MAIN OUTCOME MEASURES: Hospital utilisation: emergency department attendance, emergency admissions, emergency admissions for ambulatory sensitive conditions, outpatient attendance, elective admission and length of stay. RESULTS: By the fourth year of the Programme, there were reductions in accident and emergency (A&E) attendance (rate ratio 0.944, 95% CI 0.913 to 0.976), outpatient attendances (rate ratio 0.938, 95% CI 0.902 to 0.975) and elective admissions (rate ratio 0.921, 95% CI 0.908 to 0.935) but there was no evidence of reduced emergency admissions. The costs of the Programme were £149 per resident aged 65 and above but savings in hospital costs were only £86 per resident aged 65 and above, equivalent to a net increase in health service expenditure of £64 per resident though the Programme was nearly cost neutral if set-up costs were excluded. Holistic assessments carried out by GPs and consequent Integrated Care Management (ICM) plans were associated with increases in elective activity and costs; £126 increase in outpatient attendance and £936 in elective admission costs per holistic assessment carried out, and £576 increase in outpatient and £5858 in elective admission costs per patient receiving ICM. CONCLUSIONS: The Older People's Programme was not cost saving. Some aspects of the Programme were associated with increased costs of elective care, possibly through the identification of unmet need.

Evaluation of telephone first approach to demand management in English general practice: observational study.

Objective To evaluate a "telephone first" approach, in which all patients wanting to see a general practitioner (GP) are asked to speak to a GP on the phone before being given an appointment for a face to face consultation.Design Time series and cross sectional analysis of routine healthcare data, data from national surveys, and primary survey data.Participants 147 general practices adopting the telephone first approach compared with a 10% random sample of other practices in England.Intervention Management support for workload planning and introduction of the telephone first approach provided by two commercial companies.Main outcome measures Number of consultations, total time consulting (59 telephone first practices, no controls). Patient experience (GP Patient Survey, telephone first practices plus controls). Use and costs of secondary care (hospital episode statistics, telephone first practices plus controls). The main analysis was intention to treat, with sensitivity analyses restricted to practices thought to be closely following the companies' protocols.Results After the introduction of the telephone first approach, face to face consultations decreased considerably (adjusted change within practices -38%, 95% confidence interval -45% to -29%; P<0.001). An average practice experienced a 12-fold increase in telephone consultations (1204%, 633% to 2290%; P<0.001). The average duration of both telephone and face to face consultations decreased, but there was an overall increase of 8% in the mean time spent consulting by GPs, albeit with large uncertainty on this estimate (95% confidence interval -1% to 17%; P=0.088). These average workload figures mask wide variation between practices, with some practices experiencing a substantial reduction in workload and others a large increase. Compared with other English practices in the national GP Patient Survey, in practices using the telephone first approach there was a large (20.0 percentage points, 95% confidence interval 18.2 to 21.9; P<0.001) improvement in length of time to be seen. In contrast, other scores on the GP Patient Survey were slightly more negative. Introduction of the telephone first approach was followed by a small (2.0%) increase in hospital admissions (95% confidence interval 1% to 3%; P=0.006), no initial change in emergency department attendance, but a small (2% per year) decrease in the subsequent rate of rise of emergency department attendance (1% to 3%; P=0.005). There was a small net increase in secondary care costs.Conclusions The telephone first approach shows that many problems in general practice can be dealt with over the phone. The approach does not suit all patients or practices and is not a panacea for meeting demand. There was no evidence to support claims that the approach would, on average, save costs or reduce use of secondary care.