Implicit self-comparisons against others could bias quality of life assessments

Objectives: To explore how patient-reported health related quality of life (HRQL) and global health status are affected by use of differing personal reference frames. We hypothesised that implicit comparisons against self at an earlier time, against healthy peers or against ill patients would greatly affect patients’ response values. Study design and setting: Patients in a randomised trial for treatment of Paget’s disease completed annual HRQL questionnaires. Supplementary questions were appended, asking the patients whether they were aware of having made implicit comparisons. Results: The majority of patients reported considering themselves a year ago (31% at baseline), themselves before becoming ill (23%) or other healthy people (24%), with similar proportions during follow up. Mean HRQL scores varied substantially according to the declared frame of reference, with differences as big as 19% of the scale score, or a standardised mean effect size of 0.74 standard deviations. Conclusion: Reported reference frames were associated with effects of similar magnitude to the differences in HRQL that are regarded as clinically important. This may be of particular concern in trials that andomise patients to management in different settings, such as treatment at home / in hospital, or surgery / chemotherapy, and might bias or obscure HRQL differences

2015 AQ Summit: Identified R&D Needs Report

The Maine Aquaculture Coordinating Council recognized these issues and identified the need to strengthen connectivity between key components of the Maine aquaculture research community, and to develop a much greater alignment between research capacity and industry needs with the aim of strengthening the competitiveness and sustainability of the aquaculture sector while growing the research capacity. This includes fostering integration of existing multidisciplinary research capacity and capability not traditionally aligned with aquaculture-related activities. On January 14th, 2015 the Maine Aquaculture Innovation Center, and the University of Maine Aquaculture Research Institute hosted the first annual Maine Aquaculture R&D Forum (later renamed to Summit). This was a unique opportunity for members of the industry and researchers to meet, exchange ideas, and explore innovative, cutting edge solutions for Maine’s aquaculture industry needs

ARI Brochure 2015

Aquaculture Research Institute at the University of Maine

2015 AQ Summit: Short Report

The Maine Aquaculture Coordinating Council recognized these issues and identified the need to strengthen connectivity between key components of the Maine aquaculture research community, and to develop a much greater alignment between research capacity and industry needs with the aim of strengthening the competitiveness and sustainability of the aquaculture sector while growing the research capacity. This includes fostering integration of existing multidisciplinary research capacity and capability not traditionally aligned with aquaculture-related activities. On January 14th, 2015 the Maine Aquaculture Innovation Center, and the University of Maine Aquaculture Research Institute hosted the first annual Maine Aquaculture R&D Forum (later renamed to Summit). This was a unique opportunity for members of the industry and researchers to meet, exchange ideas, and explore innovative, cutting edge solutions for Maine’s aquaculture industry needs

Maine’s Aquaculture Sector & Its R&D Priorities

Within a competitive world economy, Maine’s economic prosperity is dependent on its geography, physical resources and human capital. In this context, Maine’s coastline and marine resources represent a unique asset supporting a wide spectrum of interlinked sectors and within this spectrum the aquaculture sector plays a major role. Of the approximate 107 aquaculture businesses in production in Maine in 2014, 71 replied to the 2015 Maine Aquaculture Economic Impact Survey. In order to provide current insights on the nature of Maine’s industry, the study aims to provide an up-to-date and accurate understanding of the economic impact aquaculture has on the state of Maine, and to determine aquaculture business owner and farm demographics

Meaning behind measurement : self-comparisons affect responses to health related quality of life questionnaires

Purpose The subjective nature of quality of life is particularly pertinent to the domain of health-related quality of life (HRQOL) research. The extent to which participants’ responses are affected by subjective information and personal reference frames is unknown. This study investigated how an elderly population living with a chronic metabolic bone disorder evaluated self-reported quality of life. Methods Participants (n = 1,331) in a multi-centre randomised controlled trial for the treatment of Paget’s disease completed annual HRQOL questionnaires, including the SF-36, EQ-5D and HAQ. Supplementary questions were added to reveal implicit reference frames used when making HRQOL evaluations. Twenty-one participants (11 male, 10 female, aged 59–91 years) were interviewed retrospectively about their responses to the supplementary questions, using cognitive interviewing techniques and semi-structured topic guides. Results The interviews revealed that participants used complex and interconnected reference frames to promote response shift when making quality of life evaluations. The choice of reference frame often reflected external factors unrelated to individual health. Many participants also stated that they were unclear whether to report general or disease-related HRQOL. Conclusions It is important, especially in clinical trials, to provide instructions clarifying whether ‘quality of life’ refers to disease-related HRQOL. Information on selfcomparison reference frames is necessary for the interpretation of responses to questions about HRQOL.The Chief Scientist Office of the Scottish Government Health Directorates, The PRISM funding bodies (the Arthritis Research Campaign, the National Association for the Relief of Paget’s disease and the Alliance for Better Bone Health)Peer reviewedAuthor final versio

Web-Based Genome-Wide Association Study Identifies Two Novel Loci and a Substantial Genetic Component for Parkinson's Disease

Although the causes of Parkinson's disease (PD) are thought to be primarily environmental, recent studies suggest that a number of genes influence susceptibility. Using targeted case recruitment and online survey instruments, we conducted the largest case-control genome-wide association study (GWAS) of PD based on a single collection of individuals to date (3,426 cases and 29,624 controls). We discovered two novel, genome-wide significant associations with PD–rs6812193 near SCARB2 (, ) and rs11868035 near SREBF1/RAI1 (, )—both replicated in an independent cohort. We also replicated 20 previously discovered genetic associations (including LRRK2, GBA, SNCA, MAPT, GAK, and the HLA region), providing support for our novel study design. Relying on a recently proposed method based on genome-wide sharing estimates between distantly related individuals, we estimated the heritability of PD to be at least 0.27. Finally, using sparse regression techniques, we constructed predictive models that account for 6%–7% of the total variance in liability and that suggest the presence of true associations just beyond genome-wide significance, as confirmed through both internal and external cross-validation. These results indicate a substantial, but by no means total, contribution of genetics underlying susceptibility to both early-onset and late-onset PD, suggesting that, despite the novel associations discovered here and elsewhere, the majority of the genetic component for Parkinson's disease remains to be discovered

A centralised public information resource for randomised trials: a scoping study to explore desirability and feasibility

BACKGROUND: There are currently several concerns about the ways in which people are recruited to participate in randomised controlled trials, the low acceptance rates among people invited to participate, and the experiences of trial participants. An information resource about on-going clinical trials designed for potential and current participants could help overcome some of these problems. METHODS: We carried out a scoping exercise to explore the desirability and feasibility of establishing such a resource. We sought the views of a range of people including people who were considering taking part in a trial, current trial participants, people who had been asked but refused to participate in a trial, consumer group representatives and researchers who design and conduct trials. RESULTS: There was broad-based support for the concept of a centralised information resource for members of the public about on-going and recently completed clinical trials. Such an information resource could be based on a database containing standardised information for each trial relating to the purpose of the trial; the interventions being compared; the implications of participation for participants; and features indicative of scientific quality and ethical probity. The usefulness of the database could be enhanced if its search facility could allow people to enter criteria such as a disease and geographic area and be presented with all the trials relevant to them, and if optional display formats could allow them to view information in varying levels of detail. Access via the Internet was considered desirable, with complementary supported access via health information services. The development of such a resource is technically feasible, but the collation of the required information would take a significant investment of resources. CONCLUSION: A centralised participant oriented information resource about clinical trials could serve several purposes. A more detailed investigation of its feasibility and exploration of its potential impacts are required

Protocol for stage 1 of the GaP study (Genetic testing acceptability for Paget's disease of bone): an interview study about genetic testing and preventive treatment: would relatives of people with Paget's disease want testing and treatment if they were available?

BACKGROUND: Paget's disease of bone (PDB) is characterised by focal increases in bone turnover, affecting one or more bones throughout the skeleton. This disrupts normal bone architecture and causes pain, deformity, deafness, osteoarthritis, and fractures. Genetic factors are recognised to play a role in PDB and it is now possible to carry out genetic tests for research. In view of this, it is timely to investigate the clinical potential for a programme of genetic testing and preventative treatment for people who have a family history of PDB, to prevent or delay the development of PDB. Evidence from non-genetic conditions, that have effective treatments, demonstrates that patients' beliefs may affect the acceptability and uptake of treatment. Two groups of beliefs (illness and treatment representations) are likely to be influential. Illness representations describe how people see their illness, as outlined in Leventhal's Self-Regulation Model. Treatment representations describe how people perceive potential treatment for their disease. People offered a programme of genetic testing and treatment will develop their own treatment representations based on what is offered, but the beliefs rather than the objective programme of treatment are likely to determine their willingness to participate. The Theory of Planned Behaviour is a theoretical model that predicts behaviours from people's beliefs about the consequences, social pressures and perceived control over the behaviour, including uptake of treatment. METHODS/DESIGN: This study aims to examine the acceptability of genetic testing, followed by preventative treatment, to relatives of people with PDB. We aim to interview people with Paget's disease, and their families, from the UK. Our research questions are: 1. What do individuals with Paget's disease think would influence the involvement of their relatives in a programme of genetic testing and preventative treatment? 2. What do relatives of Paget's disease sufferers think would influence them in accepting an offer of a programme of genetic testing and preventative treatment? DISCUSSION: Our research will be informed by relevant psychological theory: primarily the Self-Regulation Model and the Theory of Planned Behaviour. The results of these interviews will inform the development of a separate questionnaire-based study to explore these research questions in greater detail

Mutations in PINK1 and Parkin Impair Ubiquitination of Mitofusins in Human Fibroblasts

PINK1 and Parkin mutations cause recessive Parkinson's disease (PD). In Drosophila and SH-SY5Y cells, Parkin is recruited by PINK1 to damaged mitochondria, where it ubiquitinates Mitofusins and consequently promotes mitochondrial fission and mitophagy