Transnational Research Project Research Articles

Cardiomyocyte-stromal cell interplay modulates the pathogenesis of arrhythmogenic cardiomyopathy

Abstract Funding Acknowledgements Type of funding sources: Public grant(s) – EU funding. Main funding source(s): Transnational Research Projects on Cardiovascular Diseases Background Arrhythmogenic Cardiomyopathy (ACM) is an inherited heart disorder characterized by a high incidence of sudden death at young age. Mutations linked to ACM occur primarily in desmosomal genes (e.g. PKP2) [1]. Different cell types individually contribute to set the ACM phenotype with either functional abnormalities (cardiomyocytes; CM) [2]; or fibro-fatty substitution (cardiac mesenchymal stromal cells; cMSC) [3]. The relative contribution of the different cell type derangements to determine disease evolution and electrical instability is still unclear. Purpose We hypothesize that the use of a multicellular cardiac system, modelling the functional interaction between CM and cMSC, will allow the understanding of the relative contribution of the different cell types to the ACM phenotypes. Methods Primary cMSC (carrier of the PKP2 mutation c.2013delC) have been collected from a right ventricle biopsy sample; control cMSC obtained from a biobank. iPSC reprogrammed from the same ACM patient and the isogenic line in which the PKP2 mutation was corrected [4] were used. Cardiac cocultures (cc) were assembled by combining 85% iPSC-CM and 15% primary cMSC (as in Figure 1A), either mutated or control, in different combination (as in Figure 1B). Fibro-fatty accumulation have been evaluated by immunofluorescence analysis. Movies of contracting clusters have been collected and analysed by the MUSCLEMOTION tool [5]. Cell-cell communication was studied in silico by InterCellar analysis [6] on the single culture transcriptomes. The assessment of the paracrine interactions was performed on the cc secretome by Olink technology. Results Immunofluorescence analysis highlighted that ACM cc (cc 4) accumulate more lipids and collagen than healthy control (HC) ones (cc 1). Since intermediate fibro-fatty levels were observed in ‘mixed cc’ (cc 2 and 3), we conclude that ACM iPSC-CM are able to influence fibro-adipo-commitment of cMSC. Contraction studies showed a high propensity for cc monolayers which include ACM iPSC-CM (cc3 and cc4) to display arrhythmic events. An increase in the percentage of arrhythmic events was triggered when the ACM cMSC were replacing HC cMSC, indicating that ACM cMSC can affect iPSC-CM rhythm. The most abundant ligand-receptor couplets regarded cell-cell adhesion, cell-matrix coupling, cell differentiation and inflammation. Differential abundance between the four cc was observed mainly for secreted factors involved in cardiac fibrosis, inflammation, adipogenesis / lipid metabolism and heart failure. In particular, DLK-1, secreted only by HC CM, is validated as novel inhibitor of ACM fibro-adipose differentiation. Conclusion Overall, our novel simple multicellular ACM cell model can recapitulate different phenotypes of ACM and contributed to understand the relative contribution of the different cell types to ACM features (e.g. fibro-adipose replacement, contractile defects and proarrhythmic events).

Maye Ma Leka – Reframing Congolese-Swedish Colonial Entaglements

This article will discuss an artistic research project exploring a repressed part of Swedish colonial history by unboxing and unfolding a hidden trove of photographs and films amassed by Swedish Missionaries in Congo. The transdisciplinary and transnational research project explores how material traces of Swedish colonial history can support contemporary discourses, processes, and practices of recovery from the colonial period's devaluation of indigenous knowledge systems. By developing a participatory practice based on artistic research methods, the project contributes new perspectives on critical re-examinations and future knowledge in artistic research.

One Health surveillance of colistin-resistant Enterobacterales in Belgium and the Netherlands between 2017 and 2019.

Colistin serves as the last line of defense against multidrug resistant Gram-negative bacterial infections in both human and veterinary medicine. This study aimed to investigate the occurrence and spread of colistin-resistant Enterobacterales (ColR-E) using a One Health approach in Belgium and in the Netherlands. In a transnational research project, a total of 998 hospitalized patients, 1430 long-term care facility (LTCF) residents, 947 children attending day care centres, 1597 pigs and 1691 broilers were sampled for the presence of ColR-E in 2017 and 2018, followed by a second round twelve months later for hospitalized patients and animals. Colistin treatment incidence in livestock farms was used to determine the association between colistin use and resistance. Selective cultures and colistin minimum inhibitory concentrations (MIC) were employed to identify ColR-E. A combination of short-read and long-read sequencing was utilized to investigate the molecular characteristics of 562 colistin-resistant isolates. Core genome multi-locus sequence typing (cgMLST) was applied to examine potential transmission events. The presence of ColR-E was observed in all One Health sectors. In Dutch hospitalized patients, ColR-E proportions (11.3 and 11.8% in both measurements) were higher than in Belgian patients (4.4 and 7.9% in both measurements), while the occurrence of ColR-E in Belgian LTCF residents (10.2%) and children in day care centres (17.6%) was higher than in their Dutch counterparts (5.6% and 12.8%, respectively). Colistin use in pig farms was associated with the occurrence of colistin resistance. The percentage of pigs carrying ColR-E was 21.8 and 23.3% in Belgium and 14.6% and 8.9% in the Netherlands during both measurements. The proportion of broilers carrying ColR-E in the Netherlands (5.3 and 1.5%) was higher compared to Belgium (1.5 and 0.7%) in both measurements. mcr-harboring E. coli were detected in 17.4% (31/178) of the screened pigs from 7 Belgian pig farms. Concurrently, four human-related Enterobacter spp. isolates harbored mcr-9.1 and mcr-10 genes. The majority of colistin-resistant isolates (419/473, 88.6% E. coli; 126/166, 75.9% Klebsiella spp.; 50/75, 66.7% Enterobacter spp.) were susceptible to the critically important antibiotics (extended-spectrum cephalosporins, fluoroquinolones, carbapenems and aminoglycosides). Chromosomal colistin resistance mutations have been identified in globally prevalent high-risk clonal lineages, including E. coli ST131 (n = 17) and ST1193 (n = 4). Clonally related isolates were detected in different patients, healthy individuals and livestock animals of the same site suggesting local transmission. Clonal clustering of E. coli ST10 and K. pneumoniae ST45 was identified in different sites from both countries suggesting that these clones have the potential to spread colistin resistance through the human population or were acquired by exposure to a common (food) source. In pig farms, the continuous circulation of related isolates was observed over time. Inter-host transmission between humans and livestock animals was not detected. The findings of this study contribute to a broader understanding of ColR-E prevalence and the possible pathways of transmission, offering insights valuable to both academic research and public health policy development.

Decolonising feminist explorations of urban futures

ABSTRACT How do decolonial feminist urban imaginaries of urban futures begin to interrogate twenty-first century urban life? The urban futures signaled in this special issue highlight three dimensions of urban imaginaries. The first connects the privatization and commodification of urban infrastructures to state-based and capitalist discursive efforts to make the urban. The second concerns temporal convergences of past, present and future in visions of the urban that reproduce the recursive logics of coloniality by re-mapping the landscapes of urban inequality and dispossession through encounters with sedimentations of colonial and neocolonial formations. Third, authors take up the everyday as a site of struggle through which women’s negotiations and placemaking practices offer alternative urban imaginaries. These articles are based on papers given at the 2019 “Feminist Explorations of Urban Futures” conference organized by the transnational feminist research project, “Urbanization and Gender in the Global South: A Transformative Knowledge Network” (GenUrb).

The Feeling of Belonging and Inclusion in a European Outermost University

It is our wish to describe the development of a transnational research project, aiming to enhance the feeling of belonging and inclusion, as a way of improving student and staff’s wellbeing, particularly at a university of a European outermost region, Madeira. Even though similar studies have been carried out in other four European universities, this paper is only focused on the University of Madeira.
 The multiple case study method was used [1-3], based on documentary analysis, surveys and focus group techniques [4] for the collected data triangulation.
 Inspired in “Impact Gaps Canvas” [5] and the “Six Conditions of Systems Change” [6], the paper wishes to convey a quick view about the process and the results in terms of policy recommendations to improve the feeling of belonging, at the University of Madeira, expecting it has a positive impact on student and staff experience and quality of learning and teaching, according to the literature review.

Trapped within the logic of modernity/coloniality

AbstractBackgroundAcademic research is the site where the production and dissemination of knowledge are embedded in Eurocentric epistemologies which are posited as universal and non‐Eurocentric knowledges are devalued, dismissed, and ignored.ObjectivesThe goal was to explore the tensions that emerged between the Global North team and the Global South team, from the Global North's perspective.MethodsThis paper a collaborative autoethnography to trace how the logics' of coloniality of power structured a transnational research project focused on exploring Indigenous Women's experience of marginalization. The autoethnography involved engaging in critical reflexive auto‐interviews between the team members in the Global North, analyzing field notes and personal written reflections.ResultsAutoethnography revealed how colonial conditioning respectively shaped both—the Global North and the Global South teams' expectations of one another, and as such, how they each operationalized decoloniality in the research process.ConclusionThe Global North team focused on epistemically disengaging from coloniality but became overly concerned on meeting the expectations of the funder's temporally oriented productivity demands and ended up rearticulating coloniality's logics. The Global South was concerned with remedying the material dimensions of coloniality in their local community but became overly focused on adopting neoliberal logic models to efficiently satisfy the North's productivity expectations. Meaning, they, too, rearticulated coloniality's logics.

Innovation, Commerce and Assisted Reproductive Technology: Opportunities and Challenges

Background: The increasing demand for ART services in Australia has supported the development of a significant number of private sector commercial providers. This sector is innovative and research active, but the effects of commercial influences on research and clinical innovation have not been fully explored. Here we report on the results of a National Health and Medical Research-funded project exploring the views and experiences of professionals working in the ART field. Aims: To elucidate the views and experiences of expert stakeholders regarding research and innovation in the ART sector. Method: We interviewed 31 ART industry stakeholders including medical, nursing, counselling, management and scientific staff, owners/directors, regulators, advocates, and referring clinicians using a standardised semi-structured interview guide. Participants were asked about strengths and weaknesses of the current system and what, if any, reform is necessary. Three team members coded the data for themes relevant to research and innovation and analysed it in accordance with the principles of thematic analysis. Results: There are several positive impacts of commercial forces on research and innovation, including competition, scale, and clinicians’ financial stake in research. Commercialised services can more easily commit to large national, transnational and technology-intensive research projects, and they can translate results rapidly into practice. It is, however, difficult to perform clinical trials in the ART setting and there are incentives to implement interventions in advance of comprehensive evaluation. Clinical innovation is a crucial component of ART practice, but it is hampered by lack of a clear, agreed distinction between innovation, experimentation, and ‘add-on’ treatments; deficiencies in the formal processes used to appraise the outcomes of innovation and challenges of communication and consent. Conclusions: Professionals who have a deep understanding of research and innovation in the ART sector recognise both benefits and harms of its current commercial organisation. To harness the benefits of commercialisation, the ART industry requires a fair, sustainable, coordinated and needs based approach to research funding and a consistent and principled-based approach to clinical innovation.

Sexual and intimate citizenship in a Time of Pandemic

This paper provides an overview of a transnational research project exploring the impact of the COVID-19 pandemic, and public health responses to it, on sexual and gendered politics. It sets out a framework for rethinking sexual and intimate citizenship during the COVID-19 pandemic, and draws on examples from India, Italy, Mexico and the UK to illustrate our analysis. We examine how the pandemic has impacted on the everyday negotiation of intimacy and highlighted material inequalities that impact on the lives of women and LGBTQ+ people. We argue that the pandemic has produced new faultlines between women and different groups of LGBTQ+ people, as well as amplifying existing tensions. In addition to identifying these faultlines, we explore the cracks opened by them which might reveal possibilities for new coalitions and alliances in relation to sexual and gendered politics.

‘Ovizire · Somgu: From Where Do We Speak?’: Artistic Interventions in the Namibian Colonial Archive (2018–2020)

In a political and cultural climate where renegotiations of the shared colonial history of Germany and Namibia have gained momentum but debates about the past remain heated and tense, the artistic practice of Namibian artist Vitjitua Ndjiharine reveals new and empathic ways to engage with colonial legacies. This article discusses a selection of Ndjiharine’s artworks, part of the transnational research and exhibition project ‘Ovizire · Somgu: From Where Do We Speak’, in Hamburg and Windhoek (2018–20). Examining her creative strategies to explore the colonial photographic archive of the Hamburg Museum am Rothenbaum elucidates how her practice relates to critical scholarly investigations of the ambivalences of photographs from the colonial era. Ndjiharine’s artistic practice not only critiques German institutions, colonial modes of representation and knowledge production, but also conjures up alternative ways to reimagine obscured and entangled colonial histories. Analysing the works exhibited in Hamburg and Windhoek allows us to attend to the distinct references and frameworks evoked to refashion and reinterpret the photographed subjects and ultimately to usher in new ways of viewing and relating to the colonial past and the present.

Subject-activity approach of S.L. Rubinstein: Neo-Kantianism and Marxism

Pages history Soviet psychology often contain gaps, which are due to incomplete or inaccessible sources as well as to ideological distortions реrception and interpretation events epoch totalitarianism. Historical-psychological reconstructions, inspired these days archival and revisionist turns, as well as methodology latent change, offer other interpretative models, on one hand, overcoming established mythologems, and on other, revealing а complex, contradictory and ambiguous picture development socio-humanitarian knowledge first half 20th сеntury. Under influence globalization and transnational research projects, contemporary Russian historiography in one way or another updates its methodological tools, turns to polyparadigmatics and transdisciplinarity, and shifts from linear interpretative schemes to constructions that include marginal and non-obvious narratives and discourses along with canonical ones. In light new interpretive model, which takes into асcount historio-graphical materials related sciences as well as hidden currents Soviet culture, three methodological milestones are singled for analysis in S.L. Rubinstein’s intellectual biography: neo-Kantian, Marxist, and anthropological (existential) реriods scientific work. It is emphasized that Soviet historiography left almost no doubts concerning Marxist foundations S.L. Rubinstein’s subjectivе-асtivity арproach, but other models interpretation not only immerse Russian psychology in context epistemological twists and turns in socio-humanitarian knowledge 20th сеntury, but also problematize established ideas and call them into question. Among such problematizations is а comprehension neo-Kantian and Marxist premises S.L. Rubinstein’s doctrine. It is stated that principle creative асtivity, notion self-development and individuation subject, problem ethics and values as internal guidelines human development represent latent neo-Kantianism in intellectual biography scientist.

Transnational online research: recognising multiple contexts in Skype-to-phone interviews

This paper lies at the intersection of discussions surrounding digitally mediated research methods and transnational research projects. It contributes to the current methodological debate surrounding online interviewing by focusing on tensions and affordances involved in Skype-to-phone interviewing in a transnational research context. While the Skype-to-phone facility does indeed increase further access to global participants, complex power hierarchies and ethical concerns continue to exist in relation to technological access/infrastructure, research governance regimes in different places and interpersonal research relations. We, therefore, propose that online researchers involved in transnational research projects using Skype methods move towards consideration of multiple competing constituencies and diverse social and spatial connectivities and power hierarchies in which they are researching. These social differences and spatial registers are not swept away through research conducted in a uniform virtual digital environment; rather transnational researchers must make explicit the multiple place-based contexts of their digitally mediated research, as they shape the research process in distinct ways. Thus, specific consideration must be given to ethical concerns that emanate from transnational online research.

¿Investigar de forma diferente? Poner en práctica los principios de la investigación feminista

Ensuring that research is ethical and contributes to the reduction of inequalities, especially gendered, class-based and racialised power imbalances, is a central tenet of feminist research. The same, ideally, goes for peacebuilding processes. However, in spite of important and meaningful attempts to redress power imbalances, both academic research and peacebuilding work too often continue to be highly unequal endeavours. These imbalances persist between the global North and global South, but also within countries. For the most part, these imbalances are inescapable, for now at least, as both research and peacebuilding are in multiple ways entangled with broader, unequal power structures that are underpinned by patriarchy, militarism, neo-coloniality and neo-liberal capitalism. However, feminist-inspired research and peacebuilding work both do also create opportunities to analyse and question these power structures, to think and act beyond and to develop less extractive and more emancipatory alternatives and practices. This interlude examines how, as a transnational feminist research project, the researchers of this thematic volume sought to develop and foster such practices together with the communities involved in the research, and what this might mean for future research in times of the COVID-19 pandemic.

Epidemiological Influences and Requirements of Global Childhood Obesity Research

Introduction: Obesity is classified as a global epidemic and judged to be the greatest public health threat in Western countries. The tremendously increasing prevalence rates in children lead to morbidity and mortality in adults. In many countries, prevalence has doubled since the 1980s. Other countries show a continuous increase or stagnate at a very high level. Given these regional differences, this study aims to draw a global world map of childhood obesity research, including regional epidemiological characteristics, to comprehensively assess research influences and needs. Methods: In addition to established bibliometric parameters, this study uses epidemiological data to interpret metadata on childhood obesity research from the Web of Science in combination with state-of-the-art visualization methods, such as density equalizing map projections. Results: It was not until the 1990s that belated recognition of the dangerous effects of childhood obesity led to an increase in the number of publications worldwide. In addition, our findings show that countries’ study output does not correlate with epidemiologic rates of childhood obesity. In contrast, the primary driver of the research efforts on childhood obesity appears to be largely driven government funding structures. Discussion/Conclusion: The geographical differences in the epidemiological background of childhood obesity complicate the implementation of transnational research projects and cross-border prevention programs. Effective realization requires a sound scientific basis, which is facilitated by globally valid approaches. Hence, there is a need for information exchange between researchers, policy makers, and private initiatives worldwide.

Inflammation of conduction tissue in patients with arrhythmic phenotype of myocarditis

Abstract Background Manifestation of arrhythmias in otherwise normal heart suggests a possible compromise of conduction tissue. It, however, remains, usually, a supposition with limited therapeutical implications. Purpose Reporting histology of inflammation/infection of conduction tissue as a cause of arrhythmic phenotype of myocarditis. Material and methods Among 420 patients with a biopsy proven myocarditis diagnosed from 2009 to 2019, 42 presented with an arrhythmic phenotype and normal cardiac anatomy and function (LVEF >50%). Of the latter, 12 subjects (28.5%; M 9; F 3; mean age 44,75±14.9 years) had included in a left ventricular endomyocardial biopsy sections of conduction tissue (CT). CT was identified by recognition of morphological Aschoff and Monckeberg criteria and positive immunostaining for HCN4. CT inflammation was defined by ≥7 CD3+ T lymphocytes with focal necrosis of adjacent cells. Cause of CT inflammation was investigated by polymerase chain reaction (PCR) of 2 frozen endomyocardial samples, immunohistochemistry for the identified viral antigens and for Tall like receptor 4 (TLR4). Results Four pts presented with non-sustained ventricular tachycardia (nsVT), seven pts with sustained (S) VT, 1 died during hospitalization because of ventricular fibrillation (VF). Inflammatory involvement of CT was documented in all 12 pts. PCR was positive for Influenza A virus in 2 pts and HHV2 in 1 with positive CT immunostaining for related antigens. In the remaining 8 pts negative PCR for viral genomes and overexpression of TLR4 suggested an immune-mediated pathway of CT inflammation. Pts with Influenza A myocarditis and CT infection responded to tamiflu (1 cp bid for 5 days) with ECG normalization while the pt with HHV2 infection died. The 8 pts with virus-negative myocarditis and TLR4 overexpession were treated with steroids and azathioprine based on TIMIC protocol. Seven of them had no more repetitive ventricular ectopic beats at Holter of 2 weeks follow-up. Conclusions Arrhythmic phenotype of myocarditis is caused by elective inflammation/infection of CT. Molecular characterization of CT damage may bring to pharmacologic control of arrhythmias in up to 75% of cases. Figure 1 Funding Acknowledgement Type of funding source: Public grant(s) – EU funding. Main funding source(s): European Project ERA-CVD “Transnational Research Projects on Cardiovascular Diseases”

Mixed‐method social network analysis for multi‐sited transnational migration research

AbstractFollowing the recent call to combine social network analysis (SNA) approaches in the study of migrant networks and their contextualization within transnational economic, social, legal and political frameworks, in this article, I review recent studies that combine mixed‐methods SNA with observational methods in the study of transnational social networks. Such studies have enabled the collection of rich and contextualized data across multiple sites, helping to address the conundrum facing transnational migration researchers: how to combine breadth and depth in transnational data collection. Mixed‐method SNA combines quantitative and qualitative SNA methods together with in‐depth repeated interviews and observations. The article shows how mixed‐method SNA helps to address three particular challenges of doing transnational migration research: designing a transnational research project; studying transnational relationships as ‘worked at'; and capturing the dynamic and changing nature of transnational relationships as embedded within multiple environments (social, legal, economic and political) in which they take place.

Telling Transnational Histories of Women in Architecture, 1960–2015

This essay uses an emergent transnational research project — a global encyclopaedia of women in architecture — as a site for unsettling the terms, chronology, and geography of feminist histories of architecture. By locating feminist architectural history in multiple geographies and histories, feminist practice can attend to the specific geopolitics of architecture and knowledge. This project uses a crowdsourced approach, rooted in local regional reference groups and writers, to facilitate a greater range of entries, voices, and expertise. Transnational histories are generated from difference and disseminate diverse models of architectural practice and lives. Biography is a central tool for providing these counter-narratives of architecture. In this essay feminist scholarship of the 1980s on women’s lives provides a critical foundation for the current biographical turn in journalism and academia. Life writing has long been foundational to women’s history writing, but contemporary biography, with its strategies of visibility, de-canonisation, and mobilisation around gender identity, also provides an affective politics for contemporary architectural feminism. The global turn in architectural history is enriched by an intersectional lens, capable of mapping the myriad geographies and differences of women’s lives, and the precise contours of agency and oppression.

Encouraging and Enabling Lifestyles and Behaviours to Simultaneously Promote Environmental Sustainability, Health and Equity: Key Policy Messages from INHERIT.

Human consumption and activity are damaging the global ecosystem and the resources on which we rely for health, well-being and survival. The COVID-19 crisis is yet another manifestation of the urgent need to transition to more sustainable societies, further exposing the weaknesses in health systems and the injustice in our societies. It also underlines that many of the factors leading to environmental degradation, ill health and social and health inequities are interlinked. The current situation provides an unprecedented opportunity to invest in initiatives that address these common factors and encourage people to live more healthily and sustainably. Such initiatives can generate the positive feedback loops needed to change the systems and structures that shape our lives. INHERIT (January 2016–December 2019), an ambitious, multisectoral and transnational research project that involved 18 organisations across Europe, funded by the European Commission, explored such solutions. It identified, defined and analysed promising inter-sectoral policies, practices and approaches to simultaneously promote environmental sustainability, protect and promote health and contribute to health equity (the INHERIT “triple-win”) and that can encourage and enable people to live, move and consume more healthfully and sustainably. It also explored the facilitators and barriers to working across sectors and in public private cooperation. The insights were brought together in guidelines setting out how policy makers can help instigate and support local “triple-win” initiatives that influence behaviours as an approach to contributing to the change that is so urgently needed to stem environmental degradation and the interlinked threats to health and wellbeing. This article sets out this guidance, providing timely insights on how to “build back better” in the post pandemic era.

The future of evacuation drills: Assessing and enhancing evacuee performance

Evacuation drills are generally the main mechanism for improving or measuring occupant performance in emergency situations, but their effectiveness is often hard to measure, and there is limited evidence for sustained training benefits. However, innovations in technology (e.g., augmented/virtual reality, novel sensors and wearable tech) offer (when combined with new approaches to designing and delivering drills) significant opportunities for a “next generation” of evidence-based evacuation drills. In this paper, we present the findings of a recent trans-national research project; we establish the main limitations of existing drills, propose a framework for the assessment of both training and evaluation aspects of drills, make a number of recommendations, and suggest a programme of work for their implementation. The paper, therefore, provides a conceptual foundation for future work which will focus on (1) establishing an evidence-based methodology for assessing evacuation drills (and alternatives), (2) harnessing novel objective and automatable approaches to data capture/analytics in order to better characterize performance, (3), developing alternatives to the current drill model, based on emerging technologies, and (4) developing guidance for regulatory bodies on the costs and benefits of each approach for different scenarios.

Using the value creation framework to capture knowledge co-creation and pathways to impact in a transnational community of practice in autism education

ABSTRACT Although theories around Communities of Practice have gained significant ground in recent years and have become an important focus for organizational development, there is a gap in studies that investigate what members gain from participation in these communities. This paper explains how the value creation framework was implemented in a transnational research and development project in autism education by examining cycles of value creation and drawing on two types of data identified by Wenger and colleagues. The value creation framework is a theoretically driven framework to assess social learning in communities. Participants involved in the learning space were co-researchers engaged in a process of investigating, sharing and reflecting on their practice. The paper discusses the methodological challenges and strengths of using the value creation framework, with a particular focus on how insights and interactions led to subsequent changes in the practice of the participants. This work has the potential to make an important contribution to methods and analysis in assessing social learning and pathways to impact in participatory research and development projects more broadly.

Research priorities for liver glycogen storage disease: An international priority setting partnership with the James Lind Alliance.

The international liver glycogen storage disease (GSD) priority setting partnership (IGSDPSP) was established to identify the top research priorities in this area. The multiphase methodology followed the principles of the James Lind Alliance (JLA) guidebook. An international scoping survey in seven languages was distributed to patients, carers, and healthcare professionals to gather uncertainties, which were consolidated into summary questions. The existing literature was reviewed to ensure that the summary questions had not yet been answered. A second survey asked responders to prioritize these summary questions. A final shortlist of 22 questions was discussed during an international multi‐stakeholder workshop, and a consensus was reached on the top 11 priorities using an adapted nominal group technique.In the first survey, a total of 1388 questions were identified from 763 responders from 58 countries. These original uncertainties were refined into 72 summary questions for a second prioritization survey. In total 562 responders from 58 countries answered the second survey. From the second survey, the top 10 for patients, carers and healthcare professionals was identified and this shortlist of 22 questions was taken to the final workshop. During the final workshop, participants identified the worldwide top 11 research priorities for liver GSD. In addition, a top three research priorities per liver GSD subtype was identified.This unique priority setting partnership is the first international, multilingual priority setting partnership focusing on ultra‐rare diseases. This process provides a valuable resource for researchers and funding agencies to foster interdisciplinary and transnational research projects with a clear benefit for patients.