55 research outputs found
Public preferences for prioritizing preventive and curative health care interventions: a discrete choice experiment
Background Setting fair health care priorities counts among the most difficult ethical challenges our societies are facing. Objective To elicit through a discrete choice experiment the Belgian adult population’s (18–75 years; N = 750) preferences for prioritizing health care and investigate whether these preferences are different for prevention versus cure. Methods We used a Bayesian D-efficient design with partial profiles, which enables considering a large number of attributes and interaction effects. We included the following attributes: 1) type of intervention (cure vs. prevention), 2) effectiveness, 3) risk of adverse effects, 4) severity of illness, 5) link between the illness and patient’s health-related lifestyle, 6) time span between intervention and effect, and 7) patient’s age group. Results All attributes were statistically significant contributors to the social value of a health care program, with patient’s lifestyle and age being the most influential ones. Interaction effects were found, showing that prevention was preferred to cure for disease in young adults, as well as for severe and lethal disease in people of any age. However, substantial differences were found in the preferences of respondents from different age groups, with different lifestyles and different health states. Conclusions Our study suggests that according to the Belgian public, contextual factors of health gains such as patient’s age and health-related lifestyle should be considered in priority setting decisions. The studies, however, revealed substantial disagreement in opinion between different population subgroups
Priority-Setting and Personality
In a publicly fnanced health system, it is important that priority-setting refects
social values. Many studies investigate public preferences through surveys taken
from samples, but to be representative, these samples must refect value judgments
of all relevant population subgroups. In this study, we explore whether, next to better-understood sources of heterogeneity such as age, education or gender, also diferences in personality play a role in how people want to set limits to health care. We
investigate the infuence of dispositional optimism: whether someone anticipates a
good or bad future. This is an important personality dimension that has been shown
to widely reverberate into people’s lives and that can also be expected to infuence
people’s views on health care. To test our hypothesis, we asked a representative
sample of the Belgian population (N=750) to complete both the revised life orientation test and a discrete choice experiment about allocating healthcare resources,
and we investigated the relationships between both measurements. We found that
more pessimistic individuals were less supportive of using patients’ age as a selection criterion and more hesitant to invest in prevention. Since individual dispositions
are usually not part of the criteria for selecting representative samples, our fndings
point at a potential non-response bias in studies that elicit social values
Organoid cell fate dynamics in space and time
Organoids are a major new tool to study tissue renewal. However, characterizing the underlying differentiation dynamics remains challenging. Here, we developed TypeTracker, which identifies cell fates by AI-enabled cell tracking and propagating end point fates back along the branched lineage trees. Cells that ultimately migrate to the villus commit to their new type early, when still deep inside the crypt, with important consequences: (i) Secretory cells commit before terminal division, with secretory fates emerging symmetrically in sister cells. (ii) Different secretory types descend from distinct stem cell lineages rather than an omnipotent secretory progenitor. (iii) The ratio between secretory and absorptive cells is strongly affected by proliferation after commitment. (iv) Spatial patterning occurs after commitment through type-dependent cell rearrangements. This "commit-then-sort"model contrasts with the conventional conveyor belt picture, where cells differentiate by moving up the crypt-villus axis and hence raises new questions about the underlying commitment and sorting mechanisms
Missense variants in ANKRD11 cause KBG syndrome by impairment of stability or transcriptional activity of the encoded protein
Purpose
Although haploinsufficiency of ANKRD11 is among the most common genetic causes of neurodevelopmental disorders, the role of rare ANKRD11 missense variation remains unclear. We characterized clinical, molecular, and functional spectra of ANKRD11 missense variants.
Methods
We collected clinical information of individuals with ANKRD11 missense variants and evaluated phenotypic fit to KBG syndrome. We assessed pathogenicity of variants through in silico analyses and cell-based experiments.
Results
We identified 20 unique, mostly de novo, ANKRD11 missense variants in 29 individuals, presenting with syndromic neurodevelopmental disorders similar to KBG syndrome caused by ANKRD11 protein truncating variants or 16q24.3 microdeletions. Missense variants significantly clustered in repression domain 2 at the ANKRD11 C-terminus. Of the 10 functionally studied missense variants, 6 reduced ANKRD11 stability. One variant caused decreased proteasome degradation and loss of ANKRD11 transcriptional activity.
Conclusion
Our study indicates that pathogenic heterozygous ANKRD11 missense variants cause the clinically recognizable KBG syndrome. Disrupted transrepression capacity and reduced protein stability each independently lead to ANKRD11 loss-of-function, consistent with haploinsufficiency. This highlights the diagnostic relevance of ANKRD11 missense variants, but also poses diagnostic challenges because the KBG-associated phenotype may be mild and inherited pathogenic ANKRD11 (missense) variants are increasingly observed, warranting stringent variant classification and careful phenotyping
Public preferences for prioritizing preventive and curative health care interventions: a discrete choice experiment
BACKGROUND: Setting fair health care priorities counts among the most difficult ethical challenges our societies are facing. OBJECTIVE: To elicit through a discrete choice experiment the Belgian adult population's (18-75 years; N = 750) preferences for prioritizing health care and investigate whether these preferences are different for prevention versus cure. METHODS: We used a Bayesian D-efficient design with partial profiles, which enables considering a large number of attributes and interaction effects. We included the following attributes: 1) type of intervention (cure vs. prevention), 2) effectiveness, 3) risk of adverse effects, 4) severity of illness, 5) link between the illness and patient's health-related lifestyle, 6) time span between intervention and effect, and 7) patient's age group. RESULTS: All attributes were statistically significant contributors to the social value of a health care program, with patient's lifestyle and age being the most influential ones. Interaction effects were found, showing that prevention was preferred to cure for disease in young adults, as well as for severe and lethal disease in people of any age. However, substantial differences were found in the preferences of respondents from different age groups, with different lifestyles and different health states. CONCLUSIONS: Our study suggests that according to the Belgian public, contextual factors of health gains such as patient's age and health-related lifestyle should be considered in priority setting decisions. The studies, however, revealed substantial disagreement in opinion between different population subgroups.publisher: Elsevier
articletitle: Public Preferences for Prioritizing Preventive and Curative Health Care Interventions: A Discrete Choice Experiment
journaltitle: Value in Health
articlelink: http://dx.doi.org/10.1016/j.jval.2014.12.007
content_type: article
copyright: Copyright © 2015 International Society for Pharmacoeconomics and Outcomes Research (ISPOR). Published by Elsevier Inc. All rights reserved.status: publishe
Priority-Setting and Personality: Effects of Dispositional Optimism on Preferences for Allocating Healthcare Resources
© 2019, Springer Science+Business Media, LLC, part of Springer Nature. In a publicly financed health system, it is important that priority-setting reflects social values. Many studies investigate public preferences through surveys taken from samples, but to be representative, these samples must reflect value judgments of all relevant population subgroups. In this study, we explore whether, next to better-understood sources of heterogeneity such as age, education or gender, also differences in personality play a role in how people want to set limits to health care. We investigate the influence of dispositional optimism: whether someone anticipates a good or bad future. This is an important personality dimension that has been shown to widely reverberate into people’s lives and that can also be expected to influence people’s views on health care. To test our hypothesis, we asked a representative sample of the Belgian population (N = 750) to complete both the revised life orientation test and a discrete choice experiment about allocating healthcare resources, and we investigated the relationships between both measurements. We found that more pessimistic individuals were less supportive of using patients’ age as a selection criterion and more hesitant to invest in prevention. Since individual dispositions are usually not part of the criteria for selecting representative samples, our findings point at a potential non-response bias in studies that elicit social values.status: publishe
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