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1.
Soc Sci Med ; 245: 112660, 2020 Jan.
Artigo em Inglês | MEDLINE | ID: mdl-31765855

RESUMO

In the UK, life extending, end-of-life (EoL) treatments are an exception to standard cost-per-quality-adjusted life year (QALY) thresholds. This implies that greater value is placed on gaining these QALYs, than QALYs gained by the majority of other patient groups treated for anything else in the health system, even for other EoL contexts (such as quality of life (QoL) improvements alone). This paper reports a Person Trade-Off (PTO) study to test whether studies that find societal support for prioritising EoL life extensions can be explained by the severity, in terms of prospective QALYs loss, of the non-terminal comparator scenarios. Eight health scenarios were designed depicting i) QoL improvements for non-EoL temporary (T-QoL) and chronic (C-QoL) health problems and ii) QoL improvements and life extensions (LEs) for EoL health problems. Preferences were elicited from a quota sample of 901 Scottish respondents in 2016 using PTO techniques via Computer Assisted Personal Interview (CAPI). Our results indicate that there is little evidence to suggest that the severity of non-EoL comparator scenarios influence preferences for EoL treatments. Respondents do not appear to have a preference for EoL over non-EoL health gains; instead there is some indication that non-EoL health gains are preferred, particularly when compared to EoL-LE health gains. Comparing between QoL and life extending EoL scenarios, our results suggest QoL improvements are preferred to life extensions. Overall, results challenge current UK EoL policy which gives additional weight to EoL health gains, particularly EoL life extensions in the case of the National Institute for Health and Care Excellence (NICE).

2.
Eur J Health Econ ; 2019 Nov 28.
Artigo em Inglês | MEDLINE | ID: mdl-31782054

RESUMO

There is an extensive body of empirical research that focuses on the societal monetary value of a quality-adjusted life year (MVQALY). Many of these studies have found the estimates to be inversely associated with the size of the health gain, and thus not conforming to the linearity assumption imposed in the QALY model. In this study, we explore the extent to which the MVQALY varies when it is associated with different types and magnitudes of quality of life (QoL) improvements. To allow for a comprehensive assessment, we derive the MVQALY corresponding to the full spectrum of health gains defined by the EQ-5D-3L instrument. The analysis was based on a large and representative sample of the population in Spain. A discrete choice experiment and a time trade-off exercise were used to derive a value set for utilities, followed by a willingness to pay questionnaire. The data were jointly analysed using regression analyses and bootstrapping techniques. Our findings indicate that societal values for a QALY corresponding to different EQ-5D-3L health gains vary approximately between 10,000€ and 30,000€. MVQALY associated with larger improvements on QoL was found to be lower than that associated with moderate QoL gains. The potential sources of the observed non-constant MVQALY are discussed.

3.
Gac Sanit ; 2019 Sep 24.
Artigo em Espanhol | MEDLINE | ID: mdl-31558385

RESUMO

Fifteen years ago, Gaceta Sanitaria published the article entitled "What is an efficient health technology in Spain?" The growing interest in setting the price of new technologies based on the value they provide to health systems and the experience accumulated by the countries in our environment make it opportune to review what constitutes an efficient health intervention in Spain in 2020. Cost-effectiveness analysis continues to be the reference method to maximize social health outcomes with the available resources. The interpretation of its results requires establishing reference values that serve as a guide on what constitutes a reasonable value for the health care system. Efficiency thresholds must be flexible and dynamic, and they need to be updated periodically. Its application should be based on and transparency, and consider other factors that reflect social preferences. Although setting thresholds is down to political decision-makers, in Spain it could be reasonable to use thresholds of 25,000 and 60,000 Euros per QALY. However, currently, in addition to determining exact figures for the threshold, the key question is whether the Spanish National Health System is able and willing to implement a payment model based on value, towards achieving gradual financing decisions and, above all, to improve the predictability, consistency and transparency of the process.

4.
Health Econ ; 28(11): 1308-1319, 2019 11.
Artigo em Inglês | MEDLINE | ID: mdl-31496009

RESUMO

Choice-based stated preference methods, such as time trade-offs (TTOs), are used to establish health state utilities informing healthcare allocation. However, little is known about the presence of (position-dependent and precedent-dependent) sequence effects in the valuation of health states, despite techniques requiring respondents to evaluate several health states in a sequence. This paper is the first to explicitly test for the presence of sequence effects in the health domain using a new explanation based on contrast effects and preference imprecision; the implication being that randomisation cannot avoid sequence effects. Six TTO questions were designed using the EQ-5D-3L descriptive system. These were grouped into two blocks of three and within each block four sequences were used. In an online survey, 1,197 Spanish respondents answered one grouping of three TTO questions. Results indicate that sequence effects can affect preferences as utilities of health states are biased downwards if preceded by a better health state and biased upwards if preceded by a worse health state. This study informs our understanding of how context effects interact with preference elicitation methods, which is essential for interpreting survey results used to inform policy.

5.
Value Health ; 22(4): 446-452, 2019 04.
Artigo em Inglês | MEDLINE | ID: mdl-30975396

RESUMO

BACKGROUND: Paired-gamble methods have been proposed to avoid the "certainty effect" associated with standard gamble methods. OBJECTIVE: This study examines the role of starting-point effects in paired-gamble methods. In particular, it examines how the utilities so derived vary as a function of the probabilities of the stimulus lottery. METHODS: A sample of 455 members of the Spanish general population valued 9 health states via face-to-face interviews. Subjects were randomly placed into 3 subgroups, which differed in terms of the stimulus gamble's probability. Nonparametric tests and an interval regression model were used to test if utilities change when the probability distribution is modified. RESULTS: Nonparametric tests showed that the probability of a health state being considered worse than death did not differ among subgroups. Nevertheless, changes in the stimulus gamble did produce significant differences in the distribution of utilities: the higher the probability of full health in the stimulus, the higher the utility elicited. Regression estimates support the existence of starting-point effects when the utilities are obtained under expected utility. According to the prospect theory, the conclusions depend on the reference point considered. When the reference points used are death or the health state evaluated, we observe differences among these groups. Nevertheless, when full health is used, these differences disappear. CONCLUSION: This research suggests that paired-gamble methods may also be susceptible to starting-point effects. Yet the differences are small, and they disappear when the data are analyzed using prospect theory with full health as the reference point.


Assuntos
Alcoolismo/diagnóstico , Indicadores Básicos de Saúde , Nível de Saúde , Teoria da Probabilidade , Qualidade de Vida , Alcoolismo/mortalidade , Alcoolismo/psicologia , Alcoolismo/terapia , Efeitos Psicossociais da Doença , Interpretação Estatística de Dados , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Modelos Estatísticos , Preferência do Paciente , Probabilidade , Anos de Vida Ajustados por Qualidade de Vida , Fatores Socioeconômicos , Espanha/epidemiologia
6.
Eur J Health Econ ; 20(4): 559-568, 2019 Jun.
Artigo em Inglês | MEDLINE | ID: mdl-30596209

RESUMO

Previous research has shown that demographics, beliefs, and self-reported own health influence TTO values. Our hypothesis is that attitudes towards length and quality of life influence TTO values, but should no longer affect a set of related choices that are based on respondents' own TTO scores. A representative sample of 1339 respondents was asked their level of agreement to four statements relating to the importance of quality and length of life. Respondents then went on to value 4 EQ-5D 5L states using an online interactive survey and a related set of 6 pairwise health-related choice questions, set up, so that respondents should be indifferent between choice options. We explored the impact of attitudes using regression analysis for TTO values and a logit model for choices. TTO values were correlated with the attitudes and were found to have a residual impact on the choices. In particular, those respondents who preferred quality of life over length of life gave less weight to the differences in years and more weight to differences in quality of life in these choice. We conclude that although the TTO responses reflect attitudes, these attitudes continue to affect health-related choices.


Assuntos
Atitude Frente a Saúde , Comportamento de Escolha , Comportamento do Consumidor , Adolescente , Adulto , Idoso , Feminino , Envelhecimento Saudável , Humanos , Expectativa de Vida , Longevidade , Masculino , Pessoa de Meia-Idade , Qualidade de Vida/psicologia , Inquéritos e Questionários , Fatores de Tempo , Adulto Jovem
7.
Health Qual Life Outcomes ; 16(1): 148, 2018 Jul 28.
Artigo em Inglês | MEDLINE | ID: mdl-30055646

RESUMO

BACKGROUND: Most health valuation studies assume that individuals' health valuations do not depend on social comparisons. However, there is some evidence that this assumption is not satisfied in practice. This paper tests whether self-rated health by means of a Visual Analogue Scale (VAS) is related to how one perceives the health of one's contemporaries, while accounting for one's health as classified by the EQ-5D classification system. METHODS: In a large sample (n = 1500), representative of the general public, we use a VAS to rate respondents' own health and their assessment of their contemporaries' health. In addition, we directly ask them whether they perceive their health to be better, the same, or worse than their contemporaries, and we measure their own health according to the EQ-5D-5 L. RESULTS: We find a positive relationship between own health rating and contemporaries' health rating, after controlling for the respondents' own health as classified according to the EQ-5D. Furthermore, we observe a discrepancy between relative health vis-à-vis age peers as measured by an ordinal comparison and relative health as measured by a VAS. Finally, respondents, especially women, tended to overestimate the health of other people of their age. CONCLUSIONS: We provide evidence that people's own health rating is related to the perception of health of contemporaries. Our results indicate that knowledge about a respondent's perception of others' health is useful in explaining health state valuations.


Assuntos
Atitude Frente a Saúde , Autoavaliação Diagnóstica , Grupo Associado , Adulto , Idoso , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Qualidade de Vida , Autoimagem , Inquéritos e Questionários , Escala Visual Analógica , Adulto Jovem
8.
Value Health ; 21(5): 596-604, 2018 05.
Artigo em Inglês | MEDLINE | ID: mdl-29753358

RESUMO

BACKGROUND: The Spanish five-level EuroQol five-dimensional questionnaire (EQ-5D-5L) valuation study was the first to use the EuroQol Valuation Technology protocol, including composite time trade-off (C-TTO) and discrete choice experiments (DCE). In this study, its investigators noticed that some interviewers did not fully explain the C-TTO task to respondents. Evidence from a follow-up study in 2014 confirmed that when interviewers followed the protocol, the distribution of C-TTO responses widened. OBJECTIVES: To handle the data quality issues in the C-TTO responses by estimating a hybrid interval regression model to produce a Spanish EQ-5D-5L value set. METHODS: Four different models were tested. Model 0 integrated C-TTO and DCE responses in a hybrid model and models 1 to 3 altered the interpretation of the C-TTO responses: model 1 allowed for censoring of the C-TTO responses, whereas model 2 incorporated interval responses and model 3 included the interviewer-specific protocol violations. For external validation, the predictions of the four models were compared with those of the follow-up study using the Lin's concordance correlation coefficient. RESULTS: This stepwise approach to modeling C-TTO and DCE responses improved the concordance between the valuation and follow-up studies (concordance correlation coefficient: 0.948 [model 0], 0.958 [model 1], 0.952 [model 2], and 0.989 [model 3]). We recommend the estimates from model 3, because its hybrid interval regression model addresses the data quality issues found in the valuation study. CONCLUSIONS: Protocol violations may occur in any valuation study; handling them in the analysis can improve external validity. The resulting EQ-5D-5L value set (model 3) can be applied to inform Spanish health technology assessments.


Assuntos
Modelos Teóricos , Qualidade de Vida , Inquéritos e Questionários , Avaliação da Tecnologia Biomédica/métodos , Comportamento de Escolha , Confiabilidade dos Dados , Nível de Saúde , Humanos , Linguagem , Análise de Regressão
9.
Health Econ ; 27(8): 1230-1246, 2018 08.
Artigo em Inglês | MEDLINE | ID: mdl-29770524

RESUMO

Preferences elicited with matching and choice usually diverge (as characterised by preference reversals), violating a basic rationality requirement, namely, procedure invariance. We report the results of an experiment that shows that preference reversals between matching (Standard Gamble in our case) and choice are reduced when the matching task is conducted using nontransparent methods. Our results suggest that techniques based on nontransparent methods are less influenced by biases (i.e., compatibility effects) than transparent methods. We also observe that imprecision of preferences influences the degree of preference reversals. The preference reversal phenomenon is less strong in subjects with more precise preferences.


Assuntos
Comportamento de Escolha , Tomada de Decisões , Jogo de Azar , Adulto , Humanos , Modelos Psicológicos , Preferência do Paciente , Adulto Jovem
10.
Eur J Health Econ ; 19(3): 315-325, 2018 Apr.
Artigo em Inglês | MEDLINE | ID: mdl-28341905

RESUMO

This study presents evidence on the role of emotions in the monetary evaluation of health technologies, namely, drug-eluting stents (DES) in our case. It is shown that subjects who are very afraid of having to undergo an angioplasty are: (a) less sensitive to the size of the risk reduction provided by DES and (b) willing to pay more. The lack of scope sensitivity questions the normative validity of the responses of highly emotional subjects. We provide evidence of this effect using what we call the cognitive-emotional random utility model and the responses of a face-to-face, computer-assisted personal interview survey conducted in a representative sample of the Spanish general population (n = 1663).


Assuntos
Emoções , Custos de Cuidados de Saúde , Análise Custo-Benefício , Stents Farmacológicos , Humanos , Inquéritos e Questionários
11.
Health Econ ; 26 Suppl 3: 97-113, 2017 12.
Artigo em Inglês | MEDLINE | ID: mdl-29285873

RESUMO

The aim of this paper is to investigate how risk attitudes in medical decisions for others vary across health contexts. A lab experiment was designed to elicit the risk attitudes of 257 medical and nonmedical students by assigning them the role of a physician who must decide between treatments for patients. An interval regression model was used to estimate individual coefficients of relative risk aversion, and an estimation model was used to test for the effect of type of medical decision and experimental design characteristics on elicited risk aversion. We find that (a) risk attitudes vary across different health contexts, but risk aversion prevails in all of them; (b) students enrolled in health-related degrees show a higher degree of risk aversion; and (c) real rewards for third parties (patients) make subjects less risk-averse. The results underline the importance of accounting for attitudes towards risk in medical decision making.


Assuntos
Atitude , Tomada de Decisões , Medição de Risco/estatística & dados numéricos , Estudantes de Ciências da Saúde/estatística & dados numéricos , Economia Comportamental , Feminino , Humanos , Masculino , Médicos
12.
Health Econ ; 26(12): e304-e318, 2017 12.
Artigo em Inglês | MEDLINE | ID: mdl-28436139

RESUMO

We present data of a contingent valuation survey, testing the effect of evaluation mode on the monetary valuation of preventing road accidents. Half of the interviewees was asked to state their willingness to pay (WTP) to reduce the risk of having only 1 type of injury (separate evaluation, SE), and the other half of the sample was asked to state their WTP for 4 types of injuries evaluated simultaneously (joint evaluation, JE). In the SE group, we observed lack of sensitivity to scope while in the JE group WTP increased with the severity of the injury prevented. However, WTP values in this group were subject to context effects. Our results suggest that the traditional explanation of the disparity between SE and JE, namely, the so-called "evaluability," does not apply here. The paper presents new explanations based on the role of preference imprecision.


Assuntos
Acidentes de Trânsito/economia , Nível de Saúde , Ferimentos e Lesões/economia , Ferimentos e Lesões/prevenção & controle , Acidentes de Trânsito/prevenção & controle , Adulto , Idoso , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Modelos Estatísticos , Inquéritos e Questionários
13.
Med Care ; 55(7): e51-e58, 2017 07.
Artigo em Inglês | MEDLINE | ID: mdl-25521503

RESUMO

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.


Assuntos
Nível de Saúde , Qualidade de Vida/psicologia , Inquéritos e Questionários/normas , Adolescente , Adulto , Idoso , Análise Custo-Benefício , Feminino , Hispano-Americanos/psicologia , Humanos , Masculino , Pessoa de Meia-Idade , Modelos Estatísticos , Adulto Jovem
14.
Med Decis Making ; 37(3): 273-284, 2017 04.
Artigo em Inglês | MEDLINE | ID: mdl-27646566

RESUMO

There is recent interest in using discrete choice experiments (DCEs) to derive health state utility values, and results can differ from time tradeoff (TTO). Clearly, DCE is "choice based," whereas TTO is generally considered a "matching" task. We explore whether procedural adaptations to the TTO, which make the method more closely resemble a DCE, make TTO and choice converge. In particular, we test whether making the matching procedure in TTO less "transparent" to the respondent reduces disparities between TTO and DCE. We designed an interactive survey that was hosted on the Internet, and 2022 interviews were achieved in the United Kingdom in a representative sample of the population. We found a marked divergence between TTO and DCE, but this was not related to the "transparency" of the TTO procedure. We conclude that a difference in the error structure between TTO and choice and that factors other than differences in utility are affecting choices is driving the divergence. The latter has fundamental implications for the way choice data are analyzed and interpreted.


Assuntos
Interpretação Estatística de Dados , Nível de Saúde , Preferência do Paciente , Qualidade de Vida , Adolescente , Adulto , Idoso , Comportamento de Escolha , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Anos de Vida Ajustados por Qualidade de Vida , Reino Unido , Adulto Jovem
15.
Eur J Health Econ ; 18(7): 921-932, 2017 Sep.
Artigo em Inglês | MEDLINE | ID: mdl-27928743

RESUMO

We provide more evidence on the functional relationship between willingness-to-pay for risk reductions and age (the senior discount). We overcome many of the limitations of previous literature that has dealt with this issue, namely, the influence of the assumptions used in statistical models on the final results. Given our large sample size (n = 6024) we can use models that are very demanding on data. We use parametric (linear, quadratic, dummies), semi-nonparametric, and non-parametric models. We also compare the marginal and the total approach and show that they provide similar results. We also overcome one of the limitations of the total approach, that is, we include the effects of socioeconomic characteristics that are correlated with age (education and income). Our main result is that all these different approaches produce very similar results, namely, they show an inverted-U relation between the value of a statistical life (VSL) and age. Those results can hardly be attributed to problems of collinearity, omitted variables or statistical assumptions. We find a clear senior discount effect. This effect seems concentrated on those who have lower education and income levels. We also find that the value of a statistical life year (VSLY) increases with age.


Assuntos
Modelos Estatísticos , Comportamento de Redução do Risco , Valor da Vida/economia , Adolescente , Adulto , Fatores Etários , Idoso , Idoso de 80 Anos ou mais , Análise Custo-Benefício , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Modelos Econométricos , Fatores Socioeconômicos , Adulto Jovem
16.
Value Health ; 19(4): 478-86, 2016 Jun.
Artigo em Inglês | MEDLINE | ID: mdl-27325340

RESUMO

BACKGROUND: Time trade-off (TTO)-based valuation studies for the three-level version of the EuroQol five-dimensional questionnaire (EQ-5D) typically started off with a ranking task (ordering the health states by preference). This was not included in the protocol for the five-level EQ-5D (EQ-5D-5L) valuation study. OBJECTIVES: To test whether reintroducing a ranking task before the composite TTO (C-TTO) could help to reduce inconsistencies in C-TTO responses and improve the data quality. METHODS: Respondents were randomly assigned to three study arms. The control arm was the present EQ-5D-5L study protocol, without ranking. The second arm (ranking without sorting) preceded the present protocol by asking respondents to rank the target health states using physical cards. The states were then valued in random order using C-TTO. In the third arm (ranking and sorting), the ranked states remained visible through the C-TTO tasks and the order of valuation was determined by the ranking. The study used only 10 EQ-5D-5L health states. We compared the C-TTO-based inconsistent pairs of health states and ties. RESULTS: The final sample size was 196 in the control arm, 205 in the ranking without sorting arm, and 199 in the ranking and sorting arm. The percentages of ties by respondents were 15.1%, 12.5%, and 12.6% for the control arm, the ranking without sorting arm, and the ranking and sorting arm, respectively. The extra cost for adding the ranking task was about 15%. CONCLUSIONS: The benefit does not justify the effort involved in the ranking task. For this reason, the addition of the ranking task to the present EQ-5D-5L valuation protocol is not an attractive option.


Assuntos
Nível de Saúde , Qualidade de Vida , Perfil de Impacto da Doença , Adolescente , Adulto , Idoso , Análise de Variância , Avaliação da Deficiência , Feminino , Humanos , Entrevistas como Assunto , Masculino , Pessoa de Meia-Idade , Anos de Vida Ajustados por Qualidade de Vida , Espanha , Inquéritos e Questionários , Adulto Jovem
17.
Gac Sanit ; 29 Suppl 1: 76-8, 2015 Sep.
Artigo em Espanhol | MEDLINE | ID: mdl-26342412

RESUMO

Cost-benefit analyses in the field of road safety compute human costs as a key component of total costs. The present article presents two studies promoted by the Directorate-General for Traffic aimed at obtaining official values for the costs associated with fatal and non-fatal traffic injuries in Spain. We combined the contingent valuation approach and the (modified) standard gamble technique in two surveys administered to large representative samples (n1=2,020, n2=2,000) of the Spanish population. The monetary value of preventing a fatality was estimated to be 1.4 million euros. Values of 219,000 and 6,100 euros were obtained for minor and severe non-fatal injuries, respectively. These figures are comparable to those observed in neighboring countries.


Assuntos
Acidentes de Trânsito/economia , Acidentes de Trânsito/mortalidade , Acidentes de Trânsito/prevenção & controle , Adulto , Efeitos Psicossociais da Doença , Análise Custo-Benefício , Estudos Transversais , Europa (Continente) , Humanos , Espanha/epidemiologia , Valor da Vida , Ferimentos e Lesões/economia , Ferimentos e Lesões/epidemiologia , Ferimentos e Lesões/etiologia
18.
Gac. sanit. (Barc., Ed. impr.) ; 29(supl.1): 76-78, sept. 2015. tab
Artigo em Espanhol | IBECS | ID: ibc-149769

RESUMO

Los análisis coste-beneficio en el ámbito de la seguridad vial han de computar la totalidad de los costes, y dentro de ellos ocupan un lugar clave los costes humanos. En esta nota se da cuenta de dos estudios promovidos por la Dirección General de Tráfico orientados a la obtención de valores oficiales para España de los costes asociados a las víctimas mortales y no mortales de los accidentes de tráfico. Mediante la combinación del enfoque de la valoración contingente con la técnica de la lotería estándar (modificada), y con muestras amplias (n1=2020, n2=2000) representativas de la población española, se estimó en 1,4 millones de euros el valor monetario de prevenir un fallecido por accidente de tráfico. Para las lesiones graves y leves, los valores estimados fueron de 219.000 y 6100 euros, respectivamente. Las cifras obtenidas tienen un orden de magnitud similar al de los países de nuestro entorno (AU)


Cost-benefit analyses in the field of road safety compute human costs as a key component of total costs. The present article presents two studies promoted by the Directorate-General for Traffic aimed at obtaining official values for the costs associated with fatal and non-fatal traffic injuries in Spain. We combined the contingent valuation approach and the (modified) standard gamble technique in two surveys administered to large representative samples (n1=2,020, n2=2,000) of the Spanish population. The monetary value of preventing a fatality was estimated to be 1.4 million euros. Values of 219,000 and 6,100 euros were obtained for minor and severe non-fatal injuries, respectively. These figures are comparable to those observed in neighboring countries (AU)


Assuntos
Humanos , Anos Potenciais de Vida Perdidos , Mortalidade Prematura/tendências , Gestão da Segurança/organização & administração , Valor da Vida , Acidentes de Trânsito/estatística & dados numéricos , Prevenção de Acidentes/estatística & dados numéricos , Custos de Cuidados de Saúde/estatística & dados numéricos , Valores Sociais , Análise Custo-Benefício
19.
BMC Med Genomics ; 8: 2, 2015 Jan 24.
Artigo em Inglês | MEDLINE | ID: mdl-25739810

RESUMO

BACKGROUND: A clinical overlap exists between mosaic Neurofibromatosis Type 2 and sporadic Schwannomatosis conditions. In these cases a molecular analysis of tumors is recommended for a proper genetic diagnostics. This analysis is challenged by the fact that schwannomas in both conditions bear a somatic double inactivation of the NF2 gene. However, SMARCB1-associated schwannomas follow a four-hit, three-step model, in which both alleles of SMARCB1 and NF2 genes are inactivated in the tumor, with one of the steps being always the loss of a big part of chromosome 22 involving both loci. CASE PRESENTATION: Here we report a 36-year-old woman who only presented multiple subcutaneous schwannomas on her right leg. To help discriminate between both possible diagnoses, an exhaustive molecular genetic and genomic analysis was performed on two schwannomas of the patient, consisting in cDNA and DNA sequencing, MLPA, microsatellite multiplex PCR and SNP-array analyses. The loss of a big part of chromosome 22 (22q12.1q13.33) was identified in both tumors. However, this loss involved the NF2 but not the SMARCB1 locus. SNP-array analysis revealed the presence of the same deletion breakpoint in both schwannomas, indicating that this alteration was actually the first NF2 inactivating hit. In addition, a distinct NF2 point mutation in each tumor was identified, representing independent second hits. In accordance with these results, no deletions or point mutations in the SMARCB1 gene were identified. None of the mutations were present in the blood. Two of the patient's children inherited chromosome 22 deleted in schwannomas of the mother, but in its wild type form. CONCLUSIONS: These results conclusively confirm the segmental mosaic NF2 nature of the clinical phenotype presented.


Assuntos
Perna (Membro) , Técnicas de Diagnóstico Molecular , Neurilemoma/genética , Neurofibromatose 2/diagnóstico , Neurofibromatose 2/genética , Adulto , Sequência de Bases , Proteínas Cromossômicas não Histona/genética , Proteínas de Ligação a DNA/genética , Diagnóstico Diferencial , Feminino , Genes da Neurofibromatose 2 , Humanos , Repetições de Microssatélites/genética , Neurilemoma/diagnóstico , Polimorfismo de Nucleotídeo Único , Proteína SMARCB1 , Fatores de Transcrição/genética
20.
Med Decis Making ; 35(3): 305-15, 2015 04.
Artigo em Inglês | MEDLINE | ID: mdl-25009190

RESUMO

BACKGROUND: Lead time tradeoff (L-TTO) is a variant of the time tradeoff (TTO). L-TTO introduces a lead period in full health before illness onset, avoiding the need to use 2 different procedures for states better and worse than dead. To estimate utilities, additive separability is assumed. We tested to what extent violations of this assumption can bias utilities estimated with L-TTO. METHODS: A sample of 500 members of the Spanish general population evaluated 24 health states, using face-to-face interviews. A total of 188 subjects were interviewed with L-TTO and the rest with TTO. Both samples evaluated the same set of 24 health states, divided into 4 groups with 6 health states per set. Each subject evaluated 1 of the sets. A random effects regression model was fitted to our data. Only health states better than dead were included in the regression since it is in this subset where additive separability can be tested clearly. RESULTS: Utilities were higher in L-TTO in relation to TTO (on average L-TTO adds about 0.2 points to the utility of health states), suggesting that additive separability is violated. The difference between methods increased with the severity of the health state. Thus, L-TTO adds about 0.14 points to the average utility of the less severe states, 0.23 to the intermediate states, and 0.28 points to the more severe estates. CONCLUSIONS: L-TTO produced higher utilities than TTO. Health problems are perceived as less severe if a lead period in full health is added upfront, implying that there are interactions between disjointed time periods. The advantages of this method have to be compared with the cost of modeling the interaction between periods.


Assuntos
Comportamento de Escolha , Morte , Nível de Saúde , Qualidade de Vida , Atividades Cotidianas , Adulto , Atitude Frente a Saúde , Transtornos Cognitivos/economia , Transtornos Cognitivos/psicologia , Análise Custo-Benefício , Feminino , Humanos , Masculino , Pessoa de Meia-Idade , Limitação da Mobilidade , Anos de Vida Ajustados por Qualidade de Vida , Índice de Gravidade de Doença , Espanha , Fatores de Tempo
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