An economic analysis of chromosome testing in couples with children who have structural chromosome abnormalities

© 2018 Thiboonboon et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. Background Structural chromosome abnormalities can cause significant negative reproductive outcomes as they typically result in morbidity and mortality of newborns. The prevalence of structural chromosomal abnormalities in live births is at least 0.05%, of which many of them have parental origins. It is uncommon to predict structural chromosome abnormalities at birth in the first child but it is possible to prevent repeated abnormalities through screening and diagnostic programmes. This study will provide an economic analysis of the prenatal detection of these abnormalities. Methods A cost-benefit analysis using a decision analytic model was employed to compare the status quo (doing nothing) with two interventional strategies. The first strategy (Strategy I) is preconceptional screening plus amniocentesis, and the second strategy (Strategy II) is amniocentesis alone. The monetary values in Thai baht (THB) were adjusted to international dollars (I$) using purchasing power parity (PPP) (I$1 = THB 17.60 for the year 2013). The robustness of the results was tested by applying a probabilistic sensitivity analysis. Results Both diagnostic strategies can reduce approximately 10.7–11.1 births with abnormal chromosomes per 1,000 diagnosed couples. The benefit cost ratios were 1.62 for Strategy I and 1.24 for Strategy II. Net present values per 1,000 diagnoses in couples were I$464,000 for Strategy I and I$267,000 for Strategy II. The probabilistic sensitivity analysis suggested that the cost-benefit analysis was sufficiently robust, confirming that both strategies provided higher benefits than costs. Conclusions Since the benefits of both diagnostic strategies exceeded their costs, both strategies are economical–with Strategy I being more economically attractive. Strategy I is superior to Strategy II because it decreases the risk of normal children potentially dying from the amniocentesis process

Variation of health-related quality of life assessed by caregivers and patients affected by severe childhood infections.

BACKGROUND: The agreement between self-reported and proxy measures of health status in ill children is not well established. This study aimed to quantify the variation in health-related quality of life (HRQOL) derived from young patients and their carers using different instruments. METHODS: A hospital-based cross-sectional survey was conducted between August 2010 and March 2011. Children with meningitis, bacteremia, pneumonia, acute otitis media, hearing loss, chronic lung disease, epilepsy, mild mental retardation, severe mental retardation, and mental retardation combined with epilepsy, aged between five to 14 years in seven tertiary hospitals were selected for participation in this study. The Health Utilities Index Mark 2 (HUI2), and Mark 3 (HUI3), and the EuroQoL Descriptive System (EQ-5D) and Visual Analogue Scale (EQ-VAS) were applied to both paediatric patients (self-assessment) and caregivers (proxy-assessment). RESULTS: The EQ-5D scores were lowest for acute conditions such as meningitis, bacteremia, and pneumonia, whereas the HUI3 scores were lowest for most chronic conditions such as hearing loss and severe mental retardation. Comparing patient and proxy scores (n = 74), the EQ-5D exhibited high correlation (r = 0.77) while in the HUI2 and HUI3 patient and caregiver scores were moderately correlated (r = 0.58 and 0.67 respectively). The mean difference between self and proxy-assessment using the HUI2, HUI3, EQ-5D and EQ-VAS scores were 0.03, 0.05, -0.03 and -0.02, respectively. In hearing-impaired and chronic lung patients the self-rated HRQOL differed significantly from their caregivers. CONCLUSIONS: The use of caregivers as proxies for measuring HRQOL in young patients affected by pneumococcal infection and its sequelae should be employed with caution. Given the high correlation between instruments, each of the HRQOL instruments appears acceptable apart from the EQ-VAS which exhibited low correlation with the others

Comparison of Economic Evaluation Methods Across Low-income, Middle-income and High-income Countries: What are the Differences and Why?

There are marked differences in methods used for undertaking economic evaluations across low-income, middle-income, and high-income countries. We outline the most apparent dissimilarities and reflect on their underlying reasons. We randomly sampled 50 studies from each of three country income groups from a comprehensive database of 2844 economic evaluations published between January 2012 and May 2014. Data were extracted on ten methodological areas: (i) availability of guidelines; (ii) research questions; (iii) perspective; (iv) cost data collection methods; (v) cost data analysis; (vi) outcome measures; (vii) modelling techniques; (viii) cost-effectiveness thresholds; (ix) uncertainty analysis; and (x) applicability. Comparisons were made across income groups and odds ratios calculated. Contextual heterogeneity rightly drives some of the differences identified. Other differences appear less warranted and may be attributed to variation in government health sector capacity, in health economics research capacity and in expectations of funders, journals and peer reviewers. By highlighting these differences, we seek to start a debate about the underlying reasons why they have occurred and to what extent the differences are conducive for methodological advancements. We suggest a number of specific areas in which researchers working in countries of differing environments could learn from one another

A decision analytic model to investigate the cost-effectiveness of poisoning prevention practices in households with young children

Background: Systematic reviews and a network meta-analysis show home safety education with or without the provision of safety equipment is effective in promoting poison prevention behaviours in households with children. This paper compares the cost-effectiveness of home safety interventions to promote poison prevention practices. Methods: A probabilistic decision-analytic model simulates healthcare costs and benefits for a hypothetical cohort of under 5 year olds. The model compares the cost-effectiveness of home safety education, home safety inspections, provision of free or low cost safety equipment and fitting of equipment. Analyses are conducted from a UK National Health Service and Personal Social Services perspective and expressed in 2012 prices. Results: Education without safety inspection, provision or fitting of equipment was the most cost-effective strategy for promoting safe storage of medicines with an incremental cost-effectiveness ratio of £2888 (95 % credible interval (CrI) £1990–£5774) per poison case avoided or £41,330 (95%CrI £20,007–£91,534) per QALY gained compared with usual care. Compared to usual care, home safety interventions were not cost-effective in promoting safe storage of other household products. Conclusion: Education offers better value for money than more intensive but expensive strategies for preventing medicinal poisonings, but is only likely to be cost-effective at £30,000 per QALY gained for families in disadvantaged areas and for those with more than one child. There was considerable uncertainty in cost-effectiveness estimates due to paucity of evidence on model parameters. Policy makers should consider both costs and effectiveness of competing interventions to ensure efficient use of resources

Cost-utility analysis of the screening program for early oral cancer detection in Thailand

10.1371/journal.pone.0207442PLoS ONE1311e020744

Cost-utility analysis of the screening program for early oral cancer detection in Thailand.

ObjectiveTo assess the cost-utility of an oral precancer screening program compared to a no-screening program in Thailand.Materials and methodsMarkov models were performed to simulate costs and Quality Adjusted Life-Years (QALYs) of both the screening and no-screening programs in the Thai population aged over 40 years. There are four steps to the screening program in Thailand: 1) mouth self-examination (MSE); 2) visual examination by trained dental nurses (VETDN); 3) visual examination by trained dentists (VETD); and 4) visual examination by oral surgeons (VEOS). The societal perspective and lifetime horizon were applied. Variables used were derived from the pilot study of the oral precancer screening program in Roi Et province as well as through patient interviews and local and international literature reviews. Results were presented in terms of Incremental Cost-Effectiveness Ratios (ICER). Sensitivity analysis was performed to assess parameters uncertainty.ResultsThe screening program yielded higher costs (1,362 Baht) and QALYs (0.0044 years) than the no screening program, producing an ICER of 311,030 Baht per QALY gained. This indicates that the screening program is cost-ineffective in the Thai context, where the cost-effectiveness threshold is THB 160,000 per QALY gained. However, the programs will be cost-effective if the screening program are improved in one of three ways; 1) the sensitivity and specificity of MSE are more than 60%, 2) the sensitivity and specificity of VETDN are greater than 90%, or 3) the low accuracy steps like MSE or VETDN are removed from the screening program.ConclusionThe screening program is found to be cost-ineffective for oral precancer detection in Thailand. However, this study suggests 3 alternative policy options to ensure the cost-effectiveness of the program

An economic analysis of chromosome testing in couples with children who have structural chromosome abnormalities

10.1371/journal.pone.0199318PLoS ONE136e019931

A scatterplot showing the results from probabilistic sensitivity analysis.

<p>A scatterplot showing the results from probabilistic sensitivity analysis.</p

Categories of structural chromosome abnormalities (SCA) and important assumptions used in the analysis.

<p>Categories of structural chromosome abnormalities (SCA) and important assumptions used in the analysis.</p