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

Drury, Nigel E; Herd, Clare P; Biglino, Giovanni; Brown, Kate; Coats, Louise; Cumper, Michael J; Guerrero, Rafael R; Miskin, Alex; Murray, Sarah; Pender, Fraser; Rooprai, Sasha; Simpson, John; Thomson, John; Weinkauf, Jara; Wootton, Julie; Jones, Timothy J.; Cowan, Katherine

doi:10.1136/openhrt-2022-002147

Cited by 12 publications

(11 citation statements)

References 21 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Of note, the majority of participants described themselves as White. The priorities therefore represent the views of the majority, White population, which has been observed in other PSPs [ 12 , 22 ]. Although siblings were included in the workshop, we had a low number of responses from them in the survey.…”

Section: Discussionmentioning

confidence: 99%

What matters to you? Engaging with children in the James Lind Alliance Children’s Cancer Priority Setting Partnership

Aldiss,

Hart-Spencer,

Langton

et al. 2023

Res Involv Engagem

View full text Add to dashboard Cite

BackgroundPrevious priority setting exercises have sought to involve children, but in the final reporting, it is evident that few children had been engaged through the process. A primary aim in the Children’s Cancer Priority Setting Partnership was to find out from children what they want research to focus on. We report on our experience to inform methods of engagement with children in future James Lind Alliance Priority Setting Partnerships and similar exercises.MethodsWe followed the James Lind Alliance process, collecting and shortlisting questions via online surveys with adult survivors of childhood cancer, carers, and professionals, and holding a final workshop. Alongside this, a parallel process to collect and prioritise questions from children was undertaken. We created animations for parents/carers to explain the project and surveys to children, gathered questions via online surveys and held a workshop with children to identify their priorities.ResultsSixty-one children and young people with cancer and 10 siblings, aged 3–21 years, submitted 252 potential questions/topics via the surveys. Submissions were refined into 24 summary questions. These questions were discussed at a workshop with eight children; they also added more questions on topics of importance to them. Workshop participants prioritised the Top 5 questions; top priority was, ‘How can we make being in hospital a better experience for children and young people? (like having better food, internet, toys, and open visiting so other family members can be more involved in the child’s care)’. The Top 5 also included cancer prevention, treatments closer to home, early diagnosis, and emotional support. These questions were taken to the final workshop at which the Top 10 priorities were decided, all five children’s priorities were reflected in the final Top 10.ConclusionsWe have demonstrated that it is possible to successfully involve children directly in setting priorities for future research. Future priority setting exercises on topics relevant to children, should seek to include their views. The Children’s Cancer Top 10 priorities reflect the voices of children and should inform the funding of future research.

show abstract

Section: Discussionmentioning

confidence: 99%

What matters to you? Engaging with children in the James Lind Alliance Children’s Cancer Priority Setting Partnership

Aldiss,

Hart-Spencer,

Langton

et al. 2023

Res Involv Engagem

View full text Add to dashboard Cite

show abstract

“…However, 91% of surgeons would be willing to randomise patients to del Nido in a clinical trial, with the combination of del Nido and St. Thomas' blood cardioplegia having the greatest acceptability. Disease identified improving organ protection during surgery as the #1 priority for research in children [17].…”

Section: Variations In Clinical Practicementioning

confidence: 99%

“…Indeed, the recent James Lind Alliance Priority Setting Partnership in Congenital Heart Disease identified improving organ protection during surgery as the number one priority for research in children. 17 …”

Section: Protecting the Immature Myocardiummentioning

confidence: 99%

Myocardial protection in paediatric cardiac surgery: building an evidence-based strategy

Drury

2024

annals

View full text Add to dashboard Cite

Cardioplegia is fundamental to the surgical repair of congenital heart defects by protecting the heart against ischaemia-reperfusion injury, characterised by low cardiac output and troponin release in the early postoperative period. The immature myocardium exhibits structural, physiological, and metabolic differences to the adult heart, with a greater sensitivity to calcium overloadmediated injury during reperfusion. del Nido cardioplegia was specifically designed to protect the immature heart, is widely used in North America, and may provide better myocardial protection in children; however, it has not been commercially available in the United Kingdom, where most centres use St Thomas' blood cardioplegia. There are no phase III clinical trials in children to support one solution over another and this lack of evidence, combined with variations in practice, suggest the presence of clinical equipoise. The best cardioplegia solution for use in children, and the impact of age and other clinical factors, remains unknown.In this Hunterian lecture, I propose an evidence-based strategy to improve myocardial protection during cardiac surgery in children through 1) conducting multi-centre clinical trials of established techniques; 2) improving our knowledge of ischaemia-reperfusion injury in the setting of cardioplegic arrest; 3) applying this to drive innovation, moving beyond current cardioplegia solutions; 4) empowering personalised medicine, through combining clinical and genomic data, including ethnic diversity; and 5) understanding the impact of cardioplegic arrest on the late outcomes that matter to patients and their families.

show abstract

“…Children undergoing surgery with cardioplegic arrest are at risk of perioperative myocardial injury, especially those with preoperative cyanosis [5,6]. Minimising damage to organs during heart surgery to reduce the frequency and severity of complications was recently identified as the #1 priority for research in children with congenital heart disease [45]. Future studies should focus on improving myocardial protection through multi-centre trials of established techniques, e.g.…”

Section: Strengths and Limitationsmentioning

confidence: 99%

The Bilateral Remote Ischaemic Conditioning in Children (BRICC) trial: a two-centre, double-blind, randomised controlled trial in young children undergoing cardiac surgery

Drury

Doorn

Woolley

et al. 2023

Preprint

Self Cite

View full text Add to dashboard Cite

Background and Aims: Previous trials evaluating remote ischaemic preconditioning in children undergoing cardiac surgery showed mixed results. We sought to determine whether adequately delivered bilateral preconditioning is cardioprotective in young children, with or without cyanosis, undergoing surgery. Methods: Prospective, double-blind, randomised controlled trial at two UK centres. Children aged 3-36 months undergoing tetralogy of Fallot repair or ventricular septal defect closure were randomised in a 1:1 ratio to receive either bilateral preconditioning or sham intervention. Participants were followed up until hospital discharge or 30 days. The primary outcome was area under the curve for high-sensitivity troponin-T in the first 24 hours after surgery, analysed by intention-to-treat. Right atrial biopsies were obtained in selected patients. Trial registration:ISRCTN12923441. Results: Between 24 October 2016 and 8 December 2020, 120 eligible children were randomised to receive either bilateral preconditioning (n=60) or sham intervention (n=60). Participants had a median age of 7 months and 42 (35%) were female. The primary outcome, area under the curve for hs-troponin-T was higher in the preconditioning group (mean: 70.0+/-50.9microg/L/hr, n=56) than in controls (mean: 55.6+/-30.1microg/L/hr, n=58), p=0.04. Sub-group analyses did not show a differential treatment effect by oxygen saturations (pinteraction=0.25) but showed evidence of differential treatment effect by underlying defect (pinteraction=0.04). Myocardial metabolism, quantified in atrial biopsies, and secondary outcomes were not different between randomised groups. Conclusions: Bilateral remote ischemic preconditioning does not attenuate myocardial injury in children undergoing surgical repair for congenital heart defects, and there was evidence of potential harm in unstented tetralogy of Fallot.

show abstract

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

Cited by 12 publications

References 21 publications

What matters to you? Engaging with children in the James Lind Alliance Children’s Cancer Priority Setting Partnership

What matters to you? Engaging with children in the James Lind Alliance Children’s Cancer Priority Setting Partnership

Myocardial protection in paediatric cardiac surgery: building an evidence-based strategy

The Bilateral Remote Ischaemic Conditioning in Children (BRICC) trial: a two-centre, double-blind, randomised controlled trial in young children undergoing cardiac surgery

Contact Info

Product

Resources

About