Venous thromboembolism and subsequent permanent work-related disability.

Essentials The burden of venous thromboembolism (VTE) related to permanent work-related disability is unknown. In a cohort of 66 005 individuals, the risk of work-related disability after a VTE was assessed. Unprovoked VTE was associated with 52% increased risk of work-related disability. This suggests that indirect costs due to loss of work time may add to the economic burden of VTE. SUMMARY: Background The burden of venous thromboembolism (VTE) related to permanent work-related disability has never been assessed among a general population. Therefore, we aimed to estimate the risk of work-related disability in subjects with incident VTE compared with those without VTE in a population-based cohort. Methods From the Tromsø Study and the Nord-Trøndelag Health Study (HUNT), Norway, 66 005 individuals aged 20-65 years were enrolled in 1994-1997 and followed to 31 December 2008. Incident VTE events among the study participants were identified and validated, and information on work-related disability was obtained from the Norwegian National Insurance Administration database. Cox-regression models using age as time-scale and VTE as time-varying exposure were used to estimate hazard ratios (HRs) with 95% confidence intervals (CIs) adjusted for sex, body mass index, smoking, education level, marital status, history of cancer, diabetes, cardiovascular disease and self-rated general health. Results During follow-up, 384 subjects had a first VTE and 9862 participants were granted disability pension. The crude incidence rate of work-related disability after VTE was 37.5 (95% CI, 29.7-47.3) per 1000 person-years, vs. 13.5 (13.2-13.7) per 1000 person-years among those without VTE. Subjects with unprovoked VTE had a 52% higher risk of work-related disability than those without VTE (HR, 1.52; 95% CI, 1.09-2.14) after multivariable adjustment, and the association appeared to be driven by deep vein thrombosis. Conclusion VTE was associated with subsequent work-related disability in a cohort recruited from the general working-age population. Our findings suggest that indirect costs because of loss of work time may add to the economic burden of VTE.

[Using value of information analysis in decision making about applied research. The case of genetic screening for hemochromatosis in Germany]. / Die Nutzung von Informationswertanalysen in Entscheidungen über angewandte Forschung. Der Fall des genetischen Screenings auf Hämochromatose in Deutschland.

Public decision makers face demands to invest in applied research in order to accelerate the adoption of new genetic tests. However, such an investment is profitable only if the results gained from further investigations have a significant impact on health care practice. An upper limit for the value of additional information aimed at improving the basis for reimbursement decisions is given by the expected value of perfect information (EVPI). This study illustrates the significance of the concept of EVPI on the basis of a probabilistic cost-effectiveness model of screening for hereditary hemochromatosis among German men. In the present example, population-based screening can barely be recommended at threshold values of 50,000 or 100,000 Euro per life year gained and also the value of additional research which might cause this decision to be overturned is small: At the mentioned threshold values, the EVPI in the German public health care system was ca. 500,000 and 2,200,000 Euro, respectively. An analysis of EVPI by individual parameters or groups of parameters shows that additional research about adherence to preventive phlebotomy could potentially provide the highest benefit. The potential value of further research also depends on methodological assumptions regarding the decision maker's time horizon as well as on scenarios with an impact on the number of affected patients and the cost-effectiveness of screening.

Health care expenditures for Medicaid-covered males with haemophilia in the United States, 2008.

Although haemophilia is an expensive disorder, no studies have estimated health care costs for Americans with haemophilia enrolled in Medicaid as distinct from those with employer-sponsored insurance (ESI). The objective of this study is to provide information on health care utilization and expenditures for publicly insured people with haemophilia in the United States in comparison with people with haemophilia who have ESI. Data from the MarketScan Medicaid Multi-State, Commercial and Medicare Supplemental databases were used for the period 2004-2008 to identify cases of haemophilia and to estimate medical expenditures during 2008. A total of 511 Medicaid-enrolled males with haemophilia were identified, 435 of whom were enrolled in Medicaid for at least 11 months during 2008. Most people with haemophilia qualified for Medicaid based on 'disability'. Average Medicaid expenditures in 2008 were $142,987 [median, $46,737], similar to findings for people with ESI. Average costs for males with haemophilia A and an inhibitor were 3.6 times higher than those for individuals without an inhibitor. Average costs for 56 adult Medicaid enrollees with HCV or HIV infection were not statistically different from those for adults without the infection, but median costs were 1.6 times higher for those treated for blood-borne infections. Haemophilia treatment can lead to high costs for payers. Further research is needed to understand the effects of public health insurance on haemophilia care and expenditures, to evaluate treatment strategies and to implement strategies that may improve outcomes and reduce costs of care.

Healthcare expenditures for males with haemophilia and employer-sponsored insurance in the United States, 2008.

Although hemophilia has a potentially high economic impact, published estimates of health care costs for Americans with hemophilia are sparse and non-specific as to the non-bleeding complications of the disease. The objective of this study is to estimate average annual health care expenditures for people with hemophilia covered by employer-sponsored insurance, stratified according to the influence of age, type of hemophilia [A (factor VIII deficiency) versus B (factor IX)], presence of neutralizing alloantibody inhibitors and exposure to blood-borne viral infections. Data from the MarketScan Commercial and Medicare Research Databases were used for the period 2002-2008 to identify cases of hemophilia and to estimate mean and median medical expenditures during 2008. A total of 1,164 males with hemophilia were identified with continuous enrollment during 2008, 933 with hemophilia A and 231 with hemophilia B. Mean health care expenditures were $155,136 [median $73,548]. Mean costs for 30 (3%) males with an inhibitor were 5 times higher than for males without an inhibitor, approximately $697,000 [median $330,835] and $144,000 [median $73,321], respectively. Clotting factor concentrate accounted for 70%-82% of total costs. Average costs for 207 adults with HCV or HIV infection were 1.5 times higher than those for adults without infection. Hemophilia treatment is costly, particularly for individuals with neutralizing alloantibody inhibitors who require bypassing agents. Efforts to understand the cause of inhibitors are needed so that prevention strategies can be implemented and the excess costs resulting from this serious complication of hemophilia care can be avoided.

Employment impact and financial burden for families of children with fragile X syndrome: findings from the National Fragile X Survey.

BACKGROUND: The employment impact and financial burden experienced by families of children with fragile X syndrome (FXS) has not been quantified in the USA. METHOD: Using a national fragile X family survey, we analysed data on 1019 families with at least one child who had a full FXS mutation. Out-of-pocket expenditures related to fragile X were reported. We used logistic regression to examine the role of insurance, number of affected children, and number of total co-occurring conditions in predicting the financial burden and employment impact of FXS, while adjusting for race, education, marital status and other sociodemographic predictors. RESULTS: Almost half of families affected by FXS reported that they had experienced an increased financial burden and nearly 60% stated that they had had to change work hours or stop work because of FXS. Families with health insurance that met family needs were significantly less likely to report an excess financial burden. The type of insurance (private or public) was not associated with the reported financial burden. Affected children's mutation status, especially male children with the full mutation, was associated with employment impact. The total number of co-occurring conditions was associated with both financial burden and employment impact. CONCLUSIONS: Families affected by FXS experienced a significant employment impact and financial burden. Policies designed to help families with FXS need to take into consideration the dimension of co-occurring conditions.

Population screening for genetic disorders in the 21st century: evidence, economics, and ethics.

BACKGROUND: Proposals for population screening for genetic diseases require careful scrutiny by decision makers because of the potential for harms and the need to demonstrate benefits commensurate with the opportunity cost of resources expended. METHODS: We review current evidence-based processes used in the United States, the United Kingdom, and the Netherlands to assess genetic screening programs, including newborn screening programs, carrier screening, and organized cascade testing of relatives of patients with genetic syndromes. In particular, we address critical evidentiary, economic, and ethical issues that arise in the appraisal of screening tests offered to the population. Specific case studies include newborn screening for congenital adrenal hyperplasia and cystic fibrosis and adult screening for hereditary hemochromatosis. RESULTS: Organizations and countries often reach different conclusions about the suitability of screening tests for implementation on a population basis. Deciding when and how to introduce pilot screening programs is challenging. In certain cases, e.g., hereditary hemochromatosis, a consensus does not support general screening although cascade screening may be cost-effective. CONCLUSION: Genetic screening policies have often been determined by technological capability, advocacy, and medical opinion rather than through a rigorous evidence-based review process. Decision making should take into account principles of ethics and opportunity costs.

The cost effectiveness of screening newborns for congenital adrenal hyperplasia.

OBJECTIVE: To assess the cost effectiveness of newborn screening for congenital adrenal hyperplasia (CAH) in the U.S. newborn population. METHODS: We constructed a decision model to estimate the incremental cost-effectiveness ratio (ICER) of CAH screening compared to a strategy of no screening. Two types of cost effectiveness analyses (CEA) were conducted to measure ICER as net cost per life year (LY): (1) traditional CEA with sensitivity and scenario analyses, and (2) probabilistic CEA. RESULTS: ICERs for (1) base-case analysis in traditional CEA and (2) probabilistic CEA were USD 292,000 and USD 255,700 per LY saved in 2005 USD, respectively. ICERs were particularly sensitive to assumptions regarding the mortality rate for the salt wasting type of CAH, in a range from 2 to 9%. The ICERs for best-case and worst-case scenarios were USD 30,900 and USD 2.9 million per LY saved, respectively. CONCLUSIONS: Using common benchmarks for cost effectiveness, our results indicate that CAH screening would be unlikely to be considered cost effective unless assumptions favorable to screening were adopted, although it could meet economic criteria used to assess U.S. regulatory policies. A limitation is that the analysis excludes outcomes such as correct assignment of gender and quality of life.

Assessment of area of involvement in skin disease: a study using schematic figure outlines.

The ability to assess the severity of dermatoses by measurement of area of involvement is important in both clinical practice and research. Using schematic figure outlines we have shown that physicians, nurses and other groups are unable to assess area accurately, and that the degree of error is partially dependent on absolute area, but also on other factors. The pattern and magnitude of the errors varied between the groups examined, but overall was such that it calls in question the use of area indices such as the PASI (psoriasis area and severity index) score in the assessment of inflammatory dermatoses.

The politics of family planning in the Maghrib.

PIP: This article uses a comparative case study approach to relate policy outcomes in terms of family planning to the patterns of political forces observed in the 3 Maghrib states of Algeria, Morocco, and Tunisia. It is suggested that official support for a strong family planning program may be linked to recognition of the problem of low labor absorption and to concrete steps taken to counter the problem. The article discusses different vantage points for approaching the political context of family planning and distinguishes between the use of family planning as an instrument of social policy and as an instrument of economic policy. Ideological reasons for opposition to or support of family planning are then outlined. The colonial experience of the 3 states is differentiated and a chronological account of their family planning programs is provided. The political systems and leadership of the 3 countries are separately discussed in greater detail, after which the influence of elite groups on family planning programs and activities in each country is assessed. Developments in the 3 countries since 1978 are then sketched. The author concludes that the relative importance of policies toward employment and women's status in connection with support for family planning has probably varied over time, with economics playing a greater role in the 1970s. The activities of non-regime political actors were found to be very significant in formulation of population policies in Algeria and Morocco but less so in Tunisia.^ieng