Research priorities in single-ventricle heart conditions: a United Kingdom national study

Abstract

Objective To bring together stakeholders in the United Kingdom to establish national priorities for research in single ventricle heart conditions. Methods This study comprised two surveys and a workshop. The initial public online survey asked respondents for up to 3 questions they would like answered by research. Responses were classified as: unanswered, already answered, or unable to be answered by scientific research. In the follow-up survey, unanswered questions were divided into categories and respondents were asked to rank categories and questions by priority. A stakeholder workshop attended by patients, parents, healthcare professionals, researchers and charities was held to determine a final list of research priorities. Results 128 respondents posed 344 research questions, of which 271 were classified as unanswered, and after removing duplicates, 204 questions remained, divided into 20 categories. 56 (49.1%) respondents successfully ranked categories and questions in the second survey. 39 participants attended the workshop, drawing up a list of 30 research priorities across 9 priority categories: Associated co-morbidities, Brain & neurodevelopment, Exercise, Fontan failure, Heart function, Living with a single ventricle heart condition, Management of the well-functioning Fontan circulation, Surgery & perioperative care, and Transplantation, mechanical support & novel therapies. Conclusions Through a multi-stage process, we engaged a wide range of interested parties to establish a list of research priorities in single ventricle heart conditions. This provides a platform for clinicians, researchers and funders in the UK and elsewhere to address the most important questions and improve outcomes in these rare but high-impact congenital heart defects.

Research priorities in children and adults with congenital heart disease: a James Lind Alliance Priority Setting Partnership

“… 26 Some of the priority topics identified were similar to our PSP, such as mechanical circulatory support/transplantation, sudden cardiac death, late outcomes of single ventricle heart conditions, mental health and pregnancy. Finally, Drury et al 19 conducted a UK national study to identify research priorities in single ventricle heart conditions, involving patients, parents, healthcare professionals, researchers and charities. Priorities included prolonging the longevity of the Fontan circulation, brain development, heart failure, pregnancy, transplantation and use of new technology.…”

“…The project was funded by a philanthropic donation, and the funder had no influence on determining the final list of priorities. The steering group anticipated that parents may be the most engaged group, as found in a previous national prioritisation exercise 19 and noted that there are more healthcare specialists working with children with CHD than with adults. To protect potential priorities for the growing population of adults living with CHD, we, therefore, split the process into parallel child/antenatal and adult tracks at the prioritisation stage.…”

Research priorities in advanced heart failure: James Lind alliance priority setting partnership

“…Comparison with existing literature Previous PSPs have covered a variety of healthcare topics, including diabetes mellitus, inflammatory bowel disease, renal transplantation, fragility fractures and congenital heart disease. [15][16][17][18][19] Each PSP has used existing networks to engage relevant groups of patients, carers and healthcare professionals. Some diseases, such as diabetes mellitus, have large, often well-engaged patient groups, so the number of respondents was higher.…”

Health and Well-Being in Surviving Congenital Heart Disease Patients: An Umbrella Review With Synthesis of Best Evidence

“…As more CHD patients live longer, research priorities need to shift from a primary focus on how to improve survival to include research on the health and well-being of those surviving through childhood and into adulthood. Patients, their families and charities who support them have identified the need for research that assesses the extent to which CHD patients can expect to live healthy, productive lives in comparison to those without CHD, including differences in health, reproductive capacity, behaviors such as physical activity and in educational attainment and career prospects (3,4). Further ongoing initiatives (e.g., The James Lind Alliance, a United Kingdom non-profit initiative that includes patients with CHD, their carers and clinicians) are expected to identify research priorities for children and adults with CHD by bringing together patients, carers and clinicians (5).…”