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Abstract Background The active involvement of patients and the public in the design and delivery of health research has been increasingly encouraged, if not enforced. Knowledge of how this is realised in practice, especially where children and young people (CYP) are concerned, is limited, partly due to the low level of reporting of patient and public involvement (PPI) in general. The aim of this work was to assess how researchers funded by the National Institute for Health and Care Research (NIHR) report the involvement of CYP in the design and conduct of child health research to better understand the opportunities offered to CYP, and the realities of involvement in practice. Methods A participation matrix, analysis framework and accompanying tools were adapted from existing frameworks, including a child-rights informed framework, the Guidance for Reporting Involvement of Patients and the Public Checklist Short Form (GRIPP2SF), and NIHR reporting expectations. Child-focused research reports were identified from the NIHR Journals Library, including any interventional or observational study involving CYP aged 0–

Summary: Surgical treatment of pediatric congenital heart disease with tissue grafts is a lifesaving intervention. Decellularization to reduce immunogenicity of tissue grafts is an increasingly popular alternative to glutaraldehyde fixation. Here, we present a protocol to decellularize porcine right ventricular outflow tracts using a 3D printed flow chamber. We describe steps for 3D printing the flow rig, preparing porcine tissue, and using the flow rig to utilize shear forces for decellularization. We then detail procedures for characterizing the acellular scaffold.For complete details on the use and execution of this protocol, please refer to Vafaee et al.1 : Publisher’s note: Undertaking any experimental protocol requires adherence to local institutional guidelines for laboratory safety and ethics.

IntroductionThe role of the arts in health is increasingly recognised, with participatory arts-based approaches facilitating public engagement. However, little is known about men's involvement in art-based participatory research. We aimed to investigate how men who are fathers may be engaged creatively to explore experiential aspects of fathering and parenthood.MethodsFathers collaborated with an artist, sharing individual perspectives around fatherhood by telephone and email, leading up to creative representations of fatherhood. Initial conversations were prompted by images from a 2020 exhibition catalogue entitled “Masculinities” (Barbican Centre, London) inviting participants' responses to the photographic curation. The catalogue served as an artistic reference to gauge a sense of participants' creative predispositions, as well as a foundation to facilitate spontaneous dialogue about personal meanings of fatherhood. Fathers' experiences of contemporary arts varied greatly; yet all fathers confidently shared responses ranging from photographers' representation of masculinity and fatherhood and perceptions of what was excluded or privileged within this very specific curation. These discussions further led to conversations around representations of fatherhood and highlighted particular areas of interest in terms of fathers' involvement in research and public engagement. The artist provided reflections to each participant by email with links to arts resources building on the initial conversations. Two further shorter sessions followed as fathers' key messages emerged, and the final forms of their own creative expressions crystallised.ResultsThe final pieces included a musical composition around sharing vulnerability as a new father, a word cloud to represent gendered language of parenthood, an animated graphic image representing the bond between father and child, a combination of short poetic stanzas highlighting assumptions around fatherhood, an experiential photographic record of a father and a son in the early years, and a cartoon strip around emotional intelligence in parenting.DiscussionArts-based participatory engagement enabled to capture deep-rooted experiences of being a father in modern society, illuminating common cultural and intergenerational perspectives, while also tapping into unique individual experiences. The richness and diversity of these unique responses suggest that arts-based methodology can facilitate public engagement with men and lead to deep reflections on complex and socially constructed phenomena such as fathering and parenthood.

Abstract Functional implications of left ventricular (LV) morphological characterization in congenital heart disease are not widely explored. This study qualitatively and quantitatively assessed LV shape associations with a) LV function and b) thoracic aortic morphology in patients with aortic coarctation (CoA) with/without bicuspid aortic valve (BAV), and healthy controls. A statistical shape modelling framework was employed to analyse three-dimensional (3D) LV shapes from cardiac magnetic resonance (CMR) data in isolated CoA (n = 25), CoA + BAV (n = 30), isolated BAV (n = 30), and healthy controls (n = 25). Average 3D templates and deformations were computed. Correlations between shape data and CMR-derived morphometric parameters (i.e., sphericity, conicity) or global and apical strain values were assessed to elucidate possible functional implications. The relationship between LV shape features and arch architecture was also explored. The LV template was shorter and more spherical in CoA patients. Sphericity was overall associated with global and apical radial (p = 0.001, R2 = 0.09; p

Communication between clinicians and patients and communication within clinical teams is widely recognized as a tool through which improved patient outcomes can be achieved. As emerging technologies, there is a notable lack of commentary on the role of immersive virtual reality (VR) and augmented reality (AR) in enhancing medical communication. This scoping review aims to map the current landscape of literature on this topic and highlights gaps in the evidence to inform future endeavors. A comprehensive search strategy was conducted across 3 databases (PubMed, Web of Science, and Embase), yielding 1000 articles, of which 623 were individually screened for relevance. Ultimately, 22 articles were selected for inclusion and review. Similarities across the cohort of studies included small sample sizes, observational study design, use of questionnaires, and more VR studies than AR. The majority of studies found these technologies to improve medical communication, although user tolerability limitations were identified. More studies are required, presenting more robust findings, in order to draw more definitive conclusions and stronger recommendations for use of immersive VR/AR in clinical environments.

Takotsubo cardiomyopathy (TCM) or “broken heart syndrome” is a rare condition that is more common in women than men, particularly those who are postmenopausal. It mimics a myocardial infarction and psychological factors have been implicated in its etiology as well as being consequences of its presentation. As part of a public engagement project we brought together 8 women (of 12 invited) previously diagnosed with TCM to facilitate a discussion, through participation in a creative workshop-based process, about their illness experience, how they made sense of it, and the meaning it had for them in their lives, and to identify areas of unmet need. Through a range of creative activities we identified that participants had high levels of unmet need in terms of information and psychosocial support. All participants enjoyed the creative process and meeting other people with a diagnosis of TCM. The workshop overall was perceived as empowering. Exploring patient narratives during artist-facilitated workshops is one approach for providing the first steps to addressing unmet need, although the importance of ensuring psychological safety cannot be over-stated.

In congenital heart disease, patches are not tailored to patient-specific anatomies, leading to shape mismatch with likely functional implications. The design of patches through imaging and modelling may be beneficial, as it could improve clinical outcomes and reduce the costs associated with redo procedures. Whilst attention has been paid to the material of the patches used in congenital surgery, this review outlines the current knowledge on this subject and isolated experimental work that uses modelling and imaging-derived information (including 3D printing) to inform the design of the surgical patch.

Abstract Background Our aim was to comprehensively review published evidence on the association between having a congenital heart disease (CHD) compared with not, on educational attainment (i.e. not obtaining a university degree, completing secondary education, or completing any vocational training vs. obtaining/completing) in adults. Method Studies were eligible if they reported the rate, odds, or proportion of level of educational attainment in adults by whether or not they had a CHD. Result Out of 1537 articles screened, we identified 11 (N = 104,585 participants, 10,487 with CHD), 10 (N = 167,470 participants, 11,820 with CHD), and 8 (N = 150,813 participants, 9817 with CHD) studies reporting information on university education, secondary education, and vocational training, respectively in both CHD and non-CHD participants. Compared to their non-CHD peers, CHD patients were more likely not to obtain a university degree (OR = 1.38, 95% CI [1.16, 1.65]), complete secondary education (OR = 1.33, 95% CI [1.09, 1.61]) or vocational training (OR = 1.11, 95% CI [0.98, 1.26]). For all three outcomes there was evidence of between study heterogeneity, with geographical area contributing to this heterogeneity. Conclusion This systematic review identified all available published data on educational attainment in CHD patients. Despite broad inclusion criteria we identified relatively few studies that included a comparison group from the same population, and amongst those that did, few adjusted for key confounders. Pooled analyses suggest evidence of lower levels of educational attainment in patients with CHD when compared to non-CHD peers. The extent to which this may be explained by confounding factors, such as parental education, or mediated by treatments is not possible to discern from the current research literature.

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.

Two patients with bicuspid aortic valve were selected for aortic valve repair using the Ozaki procedure. Patient-specific models of their aortic roots were generated based on computed tomography data and were 3-dimensional printed using a flexible resin. The models allowed sizing of the valve leaflets and practicing of leaflet suturing. (Level of Difficulty: Advanced.)

ObjectivesEffective management of aortic coarctation (CoA) affects long-term cardiovascular outcomes. Full appreciation of CoA hemodynamics is important. This study aimed to analyze the relationship between aortic shape and hemodynamic parameters by means of computational simulations, purposely isolating the morphological variable.MethodsComputational simulations were run in three aortic models. MRI-derived aortic geometries were generated using a statistical shape modeling methodology. Starting from n = 108 patients, the mean aortic configuration was derived in patients without CoA (n = 37, “no-CoA”), with surgically repaired CoA (n = 58, “r-CoA”) and with unrepaired CoA (n = 13, “CoA”). As such, the aortic models represented average configurations for each scenario. Key hemodynamic parameters (i.e., pressure drop, aortic velocity, vorticity, wall shear stress WSS, and length and number of strong flow separations in the descending aorta) were measured in the three models at three time points (peak systole, end systole, end diastole).ResultsComparing no-CoA and CoA revealed substantial differences in all hemodynamic parameters. However, simulations revealed significant increases in vorticity at the site of CoA repair, higher WSS in the descending aorta and a 12% increase in power loss, in r-CoA compared to no-CoA, despite no clinically significant narrowing (CoA index >0.8) in the r-CoA model.ConclusionsSmall alterations in aortic morphology impact on key hemodynamic indices. This may contribute to explaining phenomena such as persistent hypertension in the absence of any clinically significant narrowing. Whilst cardiovascular events in these patients may be related to hypertension, the role of arch geometry may be a contributory factor.

Congenital heart defects (CHD) introduce haemodynamic changes; e.g., bicuspid aortic valve (BAV) presents a turbulent helical flow, which activates aortic pathological processes. Flow quantification is crucial for diagnostics and to plan corrective strategies. Multiple imaging modalities exist, with phase contrast magnetic resonance imaging (PC-MRI) being the current gold standard; however, multiple predetermined site measurements may be required, while 4D MRI allows for measurements of area (A) and velocity (U) in all spatial dimensions, acquiring a single volume and enabling a retrospective analysis at multiple locations. We assessed the feasibility of gathering hemodynamic insight into aortic hemodynamics by means of wave intensity analysis (WIA) derived from 4D MRI. Data were collected in n = 12 BAV patients and n = 7 healthy controls. Following data acquisition, WIA was successfully derived at three planes (ascending, thoracic and descending aorta) in all cases. The values of wave speed were physiological and, while the small sample limited any clinical interpretation of the results, the study shows the possibility of studying wave travel and wave reflection based on 4D MRI. Below, we demonstrate for the first time the feasibility of deriving wave intensity analysis from 4D flow data and open the door to research applications in different cardiovascular scenarios.

BackgroundVentriculo-arterial (VA) coupling in bicuspid aortic valve (BAV) patients can be affected by the global aortopathy characterizing BAV disease and the presence of concomitant congenital lesions such as aortic coarctation (COA). This study aimed to isolate the COA variable and use cardiovascular magnetic resonance (CMR) imaging to perform wave intensity analysis non-invasively to shed light on VA coupling changes in BAV. The primary hypothesis was that BAV patients with COA exhibit unfavorable VA coupling, and the secondary hypothesis was that BAV patients with COA exhibit increased wave speed as a marker of reduced aortic distensibility despite successful surgical correction.MethodsPatients were retrospectively identified from a CMR database and divided into two groups: isolated BAV and BAV associated with repaired COA. Aortic and ventricular dimensions, global longitudinal strain (GLS), and ascending aortic flow data and area were collected and used to derive wave intensity from CMR data. The main variables for the analysis included all wave magnitudes (forward compression/expansion waves, FCW and FEW, respectively, and reflected backward compression wave, BCW) and wave speed.ResultsIn the comparison of patients with isolated BAV and those with BAV associated with repaired COA (n = 25 in each group), no differences were observed in left ventricular ejection fraction, GLS, or ventricular volumes, whilst significant increases in FCW and FEW magnitude were noted in the BAV and repaired COA group. The FCW inversely correlated with age and aortic size. Whilst the BCW was not significantly different compared with that in patients with/without COA, its magnitude tends to increase with a lower COA index. Patients with repaired COA exhibited higher wave speed velocity. Aortic wave speed (inversely related to distensibility) was not significantly different between the two groups.ConclusionIn the absence of a significant restenosis, VA coupling in patients with BAV and COA is not negatively affected compared to patients with isolated BAV. A reduction in the magnitude of the early systolic FCW was observed in patients who were older and with larger aortic diameters.

Congenital heart disease (CHD) is the most predominant birth defect and can require several invasive surgeries throughout childhood. The absence of materials with growth and remodelling potential is a limitation of currently used prosthetics in cardiovascular surgery, as well as their susceptibility to calcification. The field of tissue engineering has emerged as a regenerative medicine approach aiming to develop durable scaffolds possessing the ability to grow and remodel upon implantation into the defective hearts of babies and children with CHD. Though tissue engineering has produced several synthetic scaffolds, most of them failed to be successfully translated in this life-endangering clinical scenario, and currently, biological scaffolds are the most extensively used. This review aims to thoroughly summarise the existing biological scaffolds for the treatment of paediatric CHD, categorised as homografts and xenografts, and present the preclinical and clinical studies. Fixation as well as techniques of decellularisation will be reported, highlighting the importance of these approaches for the successful implantation of biological scaffolds that avoid prosthetic rejection. Additionally, cardiac scaffolds for paediatric CHD can be implanted as acellular prostheses, or recellularised before implantation, and cellularisation techniques will be extensively discussed.

Background In the UK, undergraduate paediatric training is brief, resulting in trainees with a lower paediatric knowledge base compared with other aspects of medicine. With congenital conditions being successfully treated at childhood, adult clinicians encounter and will need to understand these complex pathologies. Patient-specific 3D printed (3DP) models have been used in clinical training, especially for rarer, complex conditions. We perform a systematic review to evaluate the evidence base in using 3DP models to train paediatricians, surgeons, medical students and nurses.Methods Online databases PubMed, Web of Science and Embase were searched between January 2010 and April 2020 using search terms relevant to “paediatrics”, “education”, “training” and “3D printing”. Participants were medical students, postgraduate trainees or clinical staff. Comparative studies (patient-specific 3DP models vs traditional teaching methods) and non-comparative studies were included. Outcomes gauged objective and subjective measures: test scores, time taken to complete tasks, self-reported confidence and personal preferences on 3DP models. If reported, the cost of and time taken to produce the models were noted.Results From 587 results, 15 studies fit the criteria of the review protocol, with 5/15 being randomised controlled studies and 10/15 focussing on cardiovascular conditions. Participants using 3DP models demonstrated improved test scores and faster times to complete procedures and identify anatomical landmarks compared with traditional teaching methods (2D diagrams, lectures, videos and supervised clinical events). User feedback was positive, reporting greater user self-confidence in understanding concepts with users wishing for integrated use of 3DP in regular teaching. Four studies reported the costs and times of production, which varied depending on model complexity and printer. 3DP models were cheaper than ‘off-the-shelf’ models available on the market and had the benefit of using real-world pathologies. These mostly non-randomised and single-centred studies did not address bias or report long-term or clinically translatable outcomes.Conclusions 3DP models were associated with greater user satisfaction and good short-term educational outcomes, with low-quality evidence. Multicentred, randomised studies with long-term follow-up and clinically assessed outcomes are needed to fully assess their benefits in this setting.PROSPERO registration number CRD42020179656.