HRB open research最新文献

Background: Rare diseases are an often chronic, progressive and life-limiting group of conditions affecting more than 30 million people in Europe. These diseases are associated with significant direct and indirect costs to a spectrum of stakeholders, ranging from individuals and their families to society overall. Further quantitative research on the economic cost for children and their families living with a rare disease is required as there is little known on this topic. This scoping review aims to document the extent and type of evidence on the economic impacts of living with a rare disease for children and their families.

Methods: This scoping review will follow the PRISMA-ScR and Joanna Briggs Institute guidelines and follow the six-stage methodology for scoping reviews: (1) identifying the research question, (2) identifying relevant studies, (3) study selection, (4) charting the data, (5) collating, summarising and reporting results and (6) knowledge user consultation. Key inclusion criteria have been developed according to the Population-Concept-Context (PCC) framework. The databases EconLit, ABI/Inform, MEDLINE, PubMed, CINAHL, and Scopus will be searched for possible articles for inclusion. Two independent reviewers will screen titles and abstracts of potential articles using a dual review process to ensure all relevant studies are included. All included articles will be assessed using a validated quality appraisal tool. A panel of patient and public involvement representatives experiencing rare diseases and knowledge users will validate the review results.

Conclusions: This scoping review will map the current literature on the economic impact of paediatric rare diseases to understand how these impacts affect children living with rare diseases and their families. This evidence has the potential to influence policy and future research in this area and will support further research on the economic impact of rare diseases on families.

Background: Diabetes is a growing global health problem. International guidelines recommend identification, screening, and referral to behavioural programmes for those at high risk of developing type 2 diabetes. Diabetes prevention programmes (DPPs) can prevent type 2 diabetes in those at high risk, however many eligible participants are not referred to these programmes. Healthcare workers (HCWs) are pivotal to the referral and recruitment processes. This study aims to identify, appraise and synthesise the evidence on barriers and facilitators to referral and recruitment to DPPs from the perspective of HCWs.

Methods: A "best fit" framework synthesis method will synthesise qualitative, quantitative, and mixed methods evidence on factors that affect HCWs referral and recruitment to DPPs, with the Theoretical Domains Framework (TDF) as the a priori framework. MEDLINE, EMBASE, CINAHL, PsychINFO, Web of Science and Scopus will be searched for primary studies published in English. Year of publication will be restricted to the last 26 years (1997-2023). Quality will be assessed using the Mixed Methods Appraisal Tool. A mix of deductive coding using the TDF and inductive coding of data that does not fit the TDF will be synthesised into themes representing the whole dataset. The relationships between the final set of themes will be explored to create a new model to understand HCWs' perspectives on referral and recruitment to DPPs. Sensitivity analysis will be carried out on this conceptual model. Confidence in the synthesised findings will be assessed using the GRADE-CERQual approach. One author will screen, extract, appraise the literature while a second author will independently verify a 20% sample at each stage.

Discussion: Participation in DPPs is key for programme impact. HCWs typically identify those at risk and refer them to DPPs. Understanding HCWs' perspectives on the barriers and facilitators to referral and recruitment will inform future implementation of DPPs.

Background: Current methods of intrapartum fetal monitoring based on heart rate, increase the rates of operative delivery but do not prevent or accurately detect fetal hypoxic brain injury. There is a need for more accurate methods of intrapartum fetal surveillance that will decrease the incidence of adverse perinatal and long-term neurodevelopmental outcomes while maintaining the lowest possible rate of obstetric intervention. Fetal pulse oximetry (FPO) is a technology that may contribute to improved intrapartum fetal wellbeing evaluation by providing a non-invasive measurement of fetal oxygenation status.

Objective: This systematic review and meta-analysis aims to synthesise the evidence examining the association between intrapartum fetal oxygen saturation levels and adverse perinatal and long-term outcomes in the offspring.

Methods: We will include randomised control trials (RCTs), cohort, cross-sectional and case-control studies which examine the use of FPO during labour as a means of measuring intrapartum fetal oxygen saturation and assess its effectiveness at detecting adverse perinatal and long-term outcomes compared to existing intrapartum surveillance methods. A detailed systematic search of PubMed, EMBASE, CINAHL, The Cochrane Library, Web of Science, ClinicalTrials.Gov and WHO ICTRP will be conducted following a detailed search strategy until February 2024. Three authors will independently review titles, abstracts and full text of articles. Two reviewers will independently extract data using a pre-defined data extraction form and assess the quality of included studies using the Risk of Bias tool for RCTs and Newcastle-Ottawa Scale for observational studies. The grading of recommendations, assessment, development, and evaluation (GRADE) approach will be used to evaluate the certainty of the evidence. We will use random-effects meta-analysis for each exposure-outcome association to calculate pooled estimates using the generic variance method. This systematic review will follow the Preferred Reporting Items for Systematic reviews and Meta-analyses and MOOSE guidelines.

Prospero registration: CRD42023457368 (04/09/2023).