Defining core outcomes of reproductive genetic carrier screening: A Delphi survey of Australian and New Zealand stakeholders

Objective

Understanding the value, benefits and harms of health interventions is needed to inform best practice and ensure responsible implementation of new approaches to patient care. Such value is demonstrated through the assessment of outcomes; however, which outcomes are assessed is often highly varied across studies and can hinder the ability to draw robust conclusions. The Core Outcome Development for Carrier Screening study aims to understand the outcomes that can meaningfully capture the value of reproductive genetic carrier screening (RGCS).

Method

The authors report an iterative, two-round online Delphi survey of Australian and New Zealand stakeholders to determine the degree of consensus regarding the core outcomes of RGCS. Panellists ranked 83 outcomes according to their perceived importance on a nine-point Likert scale. Using the distribution of rankings, outcomes were grouped into tiers representative of their perceived level of importance and agreement between groups.

Results

The top tier outcomes represent those agreed to be critically important for all future studies of RGCS to assess and were used to define a preliminary core outcome set encompassing the domains (1) primary laboratory outcomes, (2) pregnancy outcomes, (3) resource use and, (4) perceived utility of RGCS.

Conclusion

These findings can guide the selection of meaningful outcomes in studies aiming to demonstrate the value of RGCS. A future international consensus process will expand on these findings and guide the inclusion of diverse perspectives across the range of settings in which RGCS is offered.     

The Impact of Local versus National Framing on Willingness to Reduce Greenhouse Gas Emissions: A case study from central Pennsylvania

This paper explores the impact of the frame in which people interpret global warming. In 1999, 637 respondents completed a mail survey in five counties in central Pennsylvania. Half of the sample received questionnaires that framed global warming in terms of the local impacts of mitigation policies, whereas the other half received questionnaires that framed global warming in terms of the national impacts of mitigation policies. The results show a statistically significant but small difference between the local and national frames in the respondents' willingness to support government policies, as well as to take voluntary actions to mitigate climate change.     

Ontogenetic variation in cold tolerance plasticity in Drosophila: is the Bogert effect bogus?

Ontogenetic variation in plasticity is important to understanding mechanisms and patterns of thermal tolerance variation. The Bogert effect postulates that, to compensate for their inability to behaviourally thermoregulate, less-mobile life stages of ectotherms are expected to show greater plasticity of thermal tolerance than more-mobile life stages. We test this general prediction by comparing plasticity of thermal tolerance (rapid cold-hardening, RCH) between mobile adults and less-mobile larvae of 16 Drosophila species. We find an RCH response in adults of 13 species but only in larvae of four species. Thus, the Bogert effect is not as widespread as expected.     

Changing places, changing emissions: The cross-scale reliability of greenhouse gas emission inventories in the US

Greenhouse gas (GHG) emission inventories, which currently inform abatement policy discussions, are developed mostly from national scale data. Nevertheless, although the policy debate tends to take place in global and national arenas, action to abate GHG emissions is inherently within the provenance of local institutions and communities. The purpose of this paper is to examine how much information is lost by not estimating GHG emissions data at scales finer than the whole US. Such information may be critical in bridging global and local policy. Differences in the composition of GHG emission sources based on GHG emission inventories at three nested spatial scales (national, state, local) for four study sites (in Kansas, North Carolina, Ohio and Pennsylvania) are analysed, drawing upon initial results of a large collaborative study known as the 'Association of American Geographers-Global Change in Local Places (GCLP)' project. The concept of spatial sovereignty of emissions is developed to test the cross-scale reliability of emission inventories. For the test year 1990, close agreement is found in the by-gas composition of GHG emissions among national, state and local inventories. Spatial sovereignty in this case is maintained. However close agreement is not found in the by-source composition of GHG emissions among national, state and local inventories. Spatial sovereignty in this case is not maintained. Regular compilation of state and local emissions source inventories may be necessary to track important spatial and temporal deviations from national trends.     

Sailing the Shoals of Adaptive Management: The Case of Salmon in the Pacific Northwest

/ Emerging ecosystem science builds on adaptive management as an approach to dealing with salmon problems in the Pacific Northwest. Adaptive management brings scientific and democratic processes together. However, managers, the public, resource users, and scientists differ in their views on the causes of salmon decline. Managers emphasize habitat loss and over-harvest as the primary causes; commercial fishers point to habitat loss, management practices, and predators; and the public gives greatest weight to water pollution and ocean drift nets. Scientific studies of salmon often produce results that seem contradictory or unclear to the public. For adaptive management to be effective, scientists' and the public need to better understand one another's perspectives.KEY WORDS: Perception; Fishery management; Salmon; Pacific Northwest; Science     

The Impact of Local versus National Framing on Willingness to Reduce Greenhouse Gas Emissions: a case study from central Pennsylvania

This paper explores the impact of the frame in which people interpret global warming. In 1999, 637 respondents completed a mail survey in five counties in central Pennsylvania. Half of the sample received questionnaires that framed global warming in terms of the local impacts of mitigation policies, whereas the other half received questionnaires that framed global warming in terms of the national impacts of mitigation policies. The results show a statistically significant but small difference between the local and national frames in the respondents' willingness to support government policies, as well as to take voluntary actions to mitigate climate change.     

OPTIMAL LONG-TERM SCHEDULING OF STORMWATER DRAINAGE REHABILITATION1

ABSTRACT: A mathematical model is developed to optimally schedule long-term stormwater infrastructure rehabilitation activities. The model is capable of considering multiple rehabilitation projects and is driven by overall cost eensiderations. Rehabilitation activities are scheduled based on perceived reliabilities and future deterioration expected within the specified planning horizon. Future growth within the stormwater drainage basin is incorporated using chance constraints that limit the likelihood that a stormwater discharge exceeds system conveyance capacity. Model structure and development are discussed, and a hypothetical example using a drainage network is presented.     

CDM Projects under the Kyoto Protocol: A Methodology for Sustainability Assessment – Experiences from South Africa and Uruguay

Under the Clean Development Mechanism (CDM) of the Kyoto Protocol, industrialised countries may finance greenhouse gases mitigation projects in developing countries. The Kyoto Protocol explicitly requires that the CDM shall assist developing countries to achieve sustainable development. However, a clear definition of sustainability for CDM projects is still debatable. MATA-CDM (Multi-Attributive Assessment of CDM Projects) is an approach that facilitates a quantitative assessment of potential projects regarding their contribution to sustainable development. This paper presents applications of MATA-CDM in two different countries. In South Africa, the application was done mainly for academic and demonstrative purposes, whereas in Uruguay it was implemented together with the responsible Designated National Authority (DNA). The work in both countries included the selection of sustainability criteria and measurable indicators. Experts weighted the criteria using personal interviews and a multi-stakeholder workshop. This method was applied to three potential CDM projects in South Africa and one in Uruguay. Results show that under the conditions of this study, the MATA-CDM approach yet fails to yield a perfect quantitative overall sustainability assessment of CDM projects but that several findings could be useful to further develop the approach with the aim to translate the vague term sustainable development to a mainstream project level. Valuable experience was in particular collected with different stakeholder processes to perform criteria weighting.