Burden of disease and costs of aneurysmal subarachnoid haemorrhage (aSAH) in the United Kingdom

<p>Abstract</p> <p>Background</p> <p>To estimate life years and quality-adjusted life years (QALYs) lost and the economic burden of aneurysmal subarachnoid haemorrhage (aSAH) in the United Kingdom including healthcare and non-healthcare costs from a societal perspective.</p> <p>Methods</p> <p>All UK residents in 2005 with aSAH (International Classification of Diseases 10^threvision (ICD-10) code I60). Sex and age-specific abridged life tables were generated for a general population and aSAH cohorts. QALYs in each cohort were calculated adjusting the life tables with health-related quality of life (HRQL) data. Healthcare costs included hospital expenditure, cerebrovascular rehabilitation, primary care and community health and social services. Non-healthcare costs included informal care and productivity losses arising from morbidity and premature death.</p> <p>Results</p> <p>A total of 80,356 life years and 74,807 quality-adjusted life years were estimated to be lost due to aSAH in the UK in 2005. aSAH costs the National Health Service (NHS) £168.2 million annually with hospital inpatient admissions accounting for 59%, community health and social services for 18%, aSAH-related operations for 15% and cerebrovascular rehabilitation for 6% of the total NHS estimated costs. The average per patient cost for the NHS was estimated to be £23,294. The total economic burden (including informal care and using the human capital method to estimate production losses) of a SAH in the United Kingdom was estimated to be £510 million annually.</p> <p>Conclusion</p> <p>The economic and disease burden of aSAH in the United Kingdom is reported in this study. Decision-makers can use these results to complement other information when informing prevention policies in this field and to relate health care expenditures to disease categories.</p

Patient preferences for management of high blood pressure in the UK:A discrete choice experiment

Background: With a variety of potentially effective hypertension management options, it is important to determine how patients value different models of care, and the relative importance of factors in their decision-making process. Aim: To explore patient preferences for the management of hypertension in the UK. Design and setting: Online survey of patients who have hypertension in the UK including an unlabelled discrete choice experiment (DCE). Method: A DCE was developed to assess patient preferences for the management of hypertension based on four attributes: model of care, frequency of blood pressure (BP) measurement, reduction in 5-year cardiovascular risk, and costs to the NHS. A mixed logit model was used to estimate preferences, willingness-to-pay was modelled, and a scenario analysis was conducted to evaluate the impact of changes in attribute levels on the uptake of different models of care. Results: One hundred and sixty-seven participants completed the DCE (aged 61.4 years, 45.0% female, 82.0% >5 years since diagnosis). All four attributes were significant in choice (P<0.05). Reduction in 5-year cardiovascular risk was the main driver of patient preference as evidenced in the scenario and willingness-to-pay analyses. GP management was significantly preferred over self-management. Patients preferred scenarios with more frequent BP measurement, and lower costs to the NHS. Conclusion: Participants had similar preferences for GP management, pharmacist management, and telehealth, but a negative preference for self-management. When introducing new models of care for hypertension to patients, discussion of the potential benefits in terms of risk reduction should be prioritised to maximise uptake

Long-term healthcare utilization and costs of babies born after assisted reproductive technologies (ART): a record linkage study with 10-years’ follow-up in England

STUDY QUESTION:Is the long-term health care utilization of children born after ART more costly to the healthcare system in England than children born to mothers with no fertility problems? SUMMARY ANSWER:Children born after ART had significantly more general practitioner (GP) consultations and higher primary care costs up to 10 years after birth, and significantly higher hospital admission costs in the first year after birth, compared to childrenborn to mothers with no fertility problems. WHAT IS KNOWN ALREADY:There is evidence that children born after ART are at an increased risk of adverse birth outcomes and a small increased risk of rare adverse outcomes in childhood. STUDY DESIGN, SIZE, DURATION: We conducted a longitudinal study of 368 088 mother and baby pairs in England using a bespoke linked dataset. Singleton babies born 1997–2018, and their mothers, who were registered at GP practices in England contributing data to the Clinical Practice Research Datalink (CPRD), were identified through the CPRD GOLD mother–baby dataset; this data was augmentedwith further linkage to the mothers’ Human Fertilisation and Embryology Authority (HFEA) Register data. Four groups of babies were identified through the mothers’ records: a ‘fertile’ comparison group, an ‘untreated sub-fertile’ group, an ‘ovulation induction’group, and an ART group. Babies were followed-up from birth to 28 February 2021, unless censored due to loss to follow-up (e.g. leaving GP practice, emigration) or death. PARTICIPANTS/MATERIALS, SETTING, METHODS: The CPRD collects anonymized coded patient electronic health records from a network of GPs in the UK. We estimated primary care costs and hospital admission costs for babies in the four fertility groups using the CPRD GOLD data and the linked Hospital Episode Statistics (HES) Admitted Patient Care (APC) data. Linear regression was used to compare the care costs in the different groups. Inverse probability weights were generated and applied to adjust for potential bias caused by attrition due to loss to follow-up. MAIN RESULTS AND THE ROLE OF CHANCE: Children born to mothers with no fertility problems had significantly fewer consultations and lower primary care costs compared to the other groups throughout the 10-years’ follow up. Regarding hospital costs, childrenborn after ART had significantly higher hospital admission costs in the first year after birth compared to those born to motherswith no fertility problems (difference = £307 (95% CI: 153, 477)). The same pattern was observed in children born after untreated subfertility and ovulation induction. LIMITATIONS, REASONS FOR CAUTION: HFEA linkage uses non-donor data cycles only, and the introduction of consent for data use reduced the availability of HFEA records after 2009. The fertility groups were derived by augmenting HFEA data with evidence from primary care records; however, there remains some potential misclassification of exposure groups. The cost of neonatal critical care is not captured in the HES APC data, which may cause underestimation of the cost differences between the comparison group and the infertility groups. WIDER IMPLICATIONS OF THE FINDINGS: The findings can help anticipate the financial impact on the healthcare system associatedwith subfertility and ART, particularly as the demand for these treatments grows. STUDY FUNDING/COMPETING INTEREST(S): C.C. and this work were funded by a UK Medical Research Council Career Development Award [MR/L019671/1] and a UK MRC Transition Support Award [MR/W029286/1]. X.H. is an Australia National Health and Medical Research Council (NHMRC) Emerging Leadership Fellow [grant number 2009253]. The authors declare no competing interest. TRIAL REGISTRATION NUMBER: N/A

Valuation and modeling of EQ-5D-5L health states using a hybrid approach

Background: The EQ-5D instrument is the most widely used preference-based health-related quality of life questionnaire in cost-effectiveness analysis of health care technologies. Recently, a version called EQ-5D-5L with 5 levels on each dimension was developed. This manuscript explores the performance of a hybrid approach for the modeling of EQ-5D-5L valuation data. Methods: Two elicitation techniques, the composite time trade-off, and discrete choice experiments, were applied to a sample of the Spanish population (n=1000) using a computer-based questionnaire. The sampling process consisted of 2 stages: stratified sampling of geographic area, followed by systematic sampling in each area. A hybrid regression model combining composite time trade-off and discrete choice data was used to estimate the potential value sets using main effects as starting point. The comparison between the models was performed using the criteria of logical consistency, goodness of fit, and parsimony. Results: Twenty-seven participants from the 1000 were removed following the exclusion criteria. The best-fitted model included 2 significant interaction terms but resulted in marginal improvements in model fit compared to the main effects model. We therefore selected the model results with main effects as a potential value set for this methodological study, based on the parsimony criteria. The results showed that the main effects hybrid model was consistent, with a range of utility values between 1 and -0.224. Conclusion: This paper shows the feasibility of using a hybrid approach to estimate a value set for EQ-5D-5L valuation data.</p

Assessing the validity of the Long-Term Conditions Questionnaire (LTCQ) in women during pregnancy and the first year following birth

Background: The aim of this study was to validate a generic patient-reported outcome measure, the Long-Term Conditions Questionnaire (LTCQ), among pregnant and postpartum women living with a pre-existing long-term condition (LTC). Methods: Cognitive interviews were conducted with women who were currently pregnant or had given birth within the past year and living with a pre-existing LTC (n=11) and with healthcare professionals working in maternal care (n=11) to explore the acceptability of LTCQ items. An online survey was subsequently administered among women who were pregnant or had given birth within the past year and living with a pre-existing LTC (n=718). Tests of validity were performed including assessing correlations between the LTCQ and reference measures, the Well-being in Pregnancy (WiP) Questionnaire and the EuroQol EQ-5D-5L. Internal consistency was assessed using the Cronbach’s alpha statistic. Results: All LTCQ items were considered relevant and appropriate for use with women who were pregnant or had given birth within the past year. The most commonly reported LTC among the online survey sample (n=718) was a mental health condition (n=350, 48.7%) followed by joint, bone and connective tissues (n= 212, 29.5%) and gastrointestinal (n=143, 19.9%) condition. Data indicated LTCQ scores behaved in a predictable pattern, demonstrating poorer scores for women reporting a greater number of LTCs; mean (SD) scores, one LTC= 61.86 (17.8), two LTCs= 55.29 (16.0), three LTCs= 49.84 (15.52) and four LTCs= 44.94 (12.2). Poorer scores were also reported for women living with at least one mental health condition compared to those reporting no mental health condition, mean score = 66.18 (SD 16.7) v 48.64 (SD 13.3), p< 0.001 respectively. As anticipated, LTCQ scores demonstrated significant correlations in the expected direction with both the EQ-5D-5L and WiP scores. For all LTCQ items, the Cronbach’s alpha statistic was 0.93. Conclusion: Data presented here indicate that the LTCQ, which assesses living well with one or more LTC, is suitable for use among pregnant and postpartum women, from both the woman’s perspective and from the perspectives of maternity healthcare professionals. Use of the LTCQ would facilitate the identification of unmet needs within this high-risk cohort and support the exploration of how LTCs may affect women throughout the pregnancy and post-natal period. Understanding unmet needs within this cohort of women provides an opportunity to link up specialist care within maternity services and enhance personalised care

Exploring women's preferences for birth settings in England:a discrete choice experiment

<div><p>Objective</p><p>To explore pregnant women’s preferences for birth setting in England.</p><p>Design</p><p>Labelled discrete choice experiment (DCE).</p><p>Setting</p><p>Online survey.</p><p>Sample</p><p>Pregnant women recruited through social media and an online panel.</p><p>Methods</p><p>We developed a DCE to assess women’s preferences for four hypothetical birth settings based on seven attributes: reputation, continuity of care, distance from home, time to see a doctor, partner able to stay overnight, chance of straightforward birth and safety for baby. We used a mixed logit model, with setting modelled as an alternative-specific constant, and conducted a scenario analysis to evaluate the impact of changes in attribute levels on uptake of birth settings.</p><p>Main outcome measures</p><p>Women’s preferences for birth setting.</p><p>Results</p><p>257 pregnant women completed the DCE. All birth setting attributes, except ‘time to see doctor’, were significant in women’s choice (p<0.05). There was significant heterogeneity in preferences for some attributes. Changes to levels for ‘safety for the baby’ and ‘partner able to stay overnight’ were associated with larger changes from baseline uptake of birth setting. If the preferences identified were translated into the real-world context up to a third of those who reported planning birth in an obstetric unit might choose a midwifery unit assuming universal access to all settings, and knowledge of the differences between settings.</p><p>Conclusions</p><p>We found that ‘safety for the baby’, ‘chance of a straightforward birth’ and ‘can the woman’s partner stay overnight following birth’ were particularly important in women’s preferences for hypothetical birth setting. If all birth settings were available to women and they were aware of the differences between them, it is likely that more low risk women who currently plan birth in OUs might choose a midwifery unit.</p></div

EQ-5D-5L valuation project for the Spanish population: a descriptive overview and preliminary results

The latest EQ-5D instrument, EQ-5D-5L, needs new country-specific valuation studies to obtain a value set adapted to the characteristics of the updated instrument. Eight countries from Europe, North and South America and Asia have participated on pilot exercises to develop a final protocol which will be commonly used to perform the valuation studies in each country. Spain is the first country where this protocol has been introduced as part of the Valuation Project for the Spanish Population. This discussion paper reports a descriptive overview of EQ-5D-5L valuation results in the Spanish population and its preliminary results.The survey has a two-stage sample plan. The first stage concentrates on the selection of Spanish regions. The 50 Spanish regions were ordered by population size, and the first 20 regions covering 80% of the total Spanish population were selected. In the second stage, a simple random sampling strategy on each of the selected regions was conducted. The sample size on each region was calculated multiplying the total sample size (1,000) by the percentage of the population on the region respect the total population of the select 20 regions. Data will be collected between 21st May and 15th June 2012 using the final agreed protocol by the EuroQol group. Primary data collection will be conducted by a specialist survey company with a second company conducting a strict quality control process to ensure interviews and data collection of highest quality. The final survey has three blocks of questions. The first block includes patient characteristics (age, gender, socioeconomic status and so on), and a respondent valuation of own health using the EQ-5D-5L. The second block contains 10 composite time trade-offs (TTO) questions, for states better than death classic TTO is used and the “Lead Time” TTO is used for those health states considered worse than death. In this case the lead time (period in full health) is 10 years and the time in the disease is another 10 years to be comparable with the classical TTO were there are 10 years in full health and 10 years in the disease. The last block contains 7 discrete choice experiments questions, where the participant has to choose between two states. Finally some questions about the difficulties of the survey are also included. Descriptive statistics of the final sample are reported. A detailed overview of summary statistics for the health state valuations is included. Different models were explored; hybrid TTO and DC model and DC conditional logistic rescaled with TTO values were used and compared

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

© 2023 The Authors. Due to be published by BMJ. This is an open access article available under a Creative Commons licence. The published version can be accessed at the following link on the publisher’s website: https://bmjpaedsopen.bmj.com/content/7/1/e001818Objectives: Babies born between 27+0 and 31+6 weeks of gestation represent the largest group of very preterm babies requiring 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 (NNRD). 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 upon data for 28,154 very preterm babies, the annual total costs of neonatal care were estimated to be £262 million, with 95% 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 policy makers.This work is supported by the National Institute for Health Research, Health Services and Delivery Research Stream, project number 15/70/104 CRN accrual was approved by the NIHR for the period (1 August 2017 to 31 August 2018)

Digital thErapy For Improved tiNnitus carE Study (DEFINE): protocol for a randomised controlled trial

Tinnitus is a common health condition, affecting approximately 15% of the UK population. The tinnitus treatment with the strongest evidence base is Cognitive Behavioural Therapy (CBT), with standard tinnitus therapy typically augmented with education, relaxation and other techniques. Availability of CBT and conventional tinnitus therapy more broadly is limited for tinnitus sufferers. The DEFINE trial aims to assess whether smartphone-delivered tinnitus therapy, the Oto app, is as effective as current standard care, one-to-one therapist-delivered tinnitus treatment for the treatment of tinnitus in adults. The trial is registered in the ISRCTN Registry: ISRCTN99577932. DEFINE is an open-label, non-inferiority, prospective, parallel design, randomised-controlled trial. Recruitment, interventions and assessments will be remote, enabling UK-wide participant involvement. 198 participants aged 18 years or more will be recruited via social media advertisement or via primary care physicians. A screening process will identify those with tinnitus that impacts health-related quality of life, and following consent smartphone-based audiometry will be performed. Randomisation 1:1 to the Oto app or one-to-one therapist-led tinnitus therapy will be performed centrally by computer, matching groups for age, sex and hearing level. Following participant allocation, the Oto app will be provided for immediate use, or a one-to-one remote therapy appointment booked to occur within approximately 1 week, with up to 6 sessions delivered. Participant outcomes will be collected at 4,12, 26 and 52 weeks via questionnaire and phone call. The primary outcome is the change in Tinnitus Functional Index (TFI) total score measured at 26 weeks following allocation. Adverse events will be recorded. A health economic evaluation in the form of a cost-utility analysis will be performed using data from participant submitted EuroQol 5D-5L and Health Utilities Index Mark 3 scores and resource use data. Trial results will be made publicly available, including a plain English summary

Multiple imputation for patient reported outcome measures in randomised controlled trials : advantages and disadvantages of imputing at the item, subscale or composite score level

Background Missing data can introduce bias in the results of randomised controlled trials (RCTs), but are typically unavoidable in pragmatic clinical research, especially when patient reported outcome measures (PROMs) are used. Traditionally applied to the composite PROMs score of multi-item instruments, some recent research suggests that multiple imputation (MI) at the item level may be preferable under certain scenarios. This paper presents practical guidance on the choice of MI models for handling missing PROMs data based on the characteristics of the trial dataset. The comparative performance of complete cases analysis, which is commonly used in the analysis of RCTs, is also considered. Methods Realistic missing at random data were simulated using follow-up data from an RCT considering three different PROMs (Oxford Knee Score (OKS), EuroQoL 5 Dimensions 3 Levels (EQ-5D-3L), 12-item Short Form Survey (SF-12)). Data were multiply imputed at the item (using ordinal logit and predicted mean matching models), sub-scale and score level; unadjusted mean outcomes, as well as treatment effects from linear regression models were obtained for 1000 simulations. Performance was assessed by root mean square errors (RMSE) and mean absolute errors (MAE). Results Convergence problems were observed for MI at the item level. Performance generally improved with increasing sample sizes and lower percentages of missing data. Imputation at the score and subscale level outperformed imputation at the item level in small sample sizes (n ≤ 200). Imputation at the item level is more accurate for high proportions of item-nonresponse. All methods provided similar results for large sample sizes (≥500) in this particular case study. Conclusions Many factors, including the prevalence of missing data in the study, sample size, the number of items within the PROM and numbers of levels within the individual items, and planned analyses need consideration when choosing an imputation model for missing PROMs data