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. 2022 Nov;9(2):e002147.
doi: 10.1136/openhrt-2022-002147.

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

Nigel E Drury  1   2 , Clare P Herd  3 , Giovanni Biglino  4   5 , Katherine L Brown  6   7 , Louise Coats  8   9 , Michael J Cumper  10 , Rafael R Guerrero  11   12 , Alex Miskin  10 , Sarah Murray  10 , Fraser Pender  10 , Sasha Rooprai  10 , John M Simpson  13   14 , John D R Thomson  15   16 , Jara Weinkauf  10 , Julie Wootton  10 , Timothy J Jones  17   2 , Katherine Cowan  18
Affiliations

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

Nigel E Drury et al. Open Heart. 2022 Nov.

Abstract

Objective: To bring together patients, parents, charities and clinicians in a Priority Setting Partnership to establish national clinical priorities for research in children and adults with congenital heart disease.

Methods: The established James Lind Alliance methodology was used to identify and prioritise research on the management of congenital heart disease, focusing on diagnosis, treatment and outcomes. An initial open survey was used to gather potential uncertainties which were filtered, categorised, converted into summary questions and checked against current evidence. In a second survey, respondents identified the unanswered questions most important to them. At two final workshops, patients, parents, charities and healthcare professionals agreed the top 10 lists of priorities for child/antenatal and adult congenital heart disease research.

Results: 524 respondents submitted 1373 individual questions, from which 313 out of scope or duplicate questions were removed. The remaining 1060 questions were distilled into summary questions and checked against existing literature, with only three questions deemed entirely answered and removed. 250 respondents completed the child/antenatal survey (56 uncertainties) and 252 completed the adult survey (47 uncertainties). The questions ranked the highest by clinicians and non-clinicians were taken forward to consensus workshops, where two sets of top 10 research priorities were agreed.

Conclusions: Through an established and equitable process, we determined national clinical priorities for congenital heart disease research. These will be taken forward by specific working groups, a national patient and public involvement group, and through the establishment of a UK and Ireland network for collaborative, multicentre clinical trials in congenital heart disease.

Keywords: delivery of health care; heart defects, congenital; research design.

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Conflict of interest statement

Competing interests: None declared.

Figures

Figure 1
Figure 1
Flow diagram through the Priority Setting Partnership process: from submitted questions, to summary questions, to uncertainties, to priorities. CHD, congenital heart disease.
See this image and copyright information in PMC

References

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