Strong AyaStrong AyaPublications
STRONG AYA is committed to open science and open access. Here you can find our publications.
Knowledge Representation of a Multicenter Adolescent and Young Adult Cancer Infrastructure: Development of the STRONG AYA Knowledge Graph
Joshi Hogenboom, Varsha Gouthamchand, Charlotte Cairns, Silvie H. M. Janssen, Kirsty Way, Andre L. A. J. Dekker, Winette T. A. van der Graaf, Anne-Sophie Darlington, Olga Husson, Leonard Y. L. Wee, Johan van Soest, Aiara Lobo Gomes
JCO Clinical Cancer Informatics, 10, e2500177 (2026). DOI
Purpose: Rare diseases are difficult to fully capture, and regularly call for large, geographically dispersed initiatives. Such initiatives are often met with data harmonization challenges. These challenges render data incompatible and impede successful realization. The STRONG AYA project is such an initiative, specifically focusing on adolescent and young adult (AYAs) with cancer. STRONG AYA is setting up a federated data infrastructure containing data of varying format. Here, we elaborate on how we used health care–agnostic semantic web technologies to overcome such challenges. Methods: We structured the STRONG AYA case-mix and core outcome measures concepts and their properties as knowledge graphs. Having identified the corresponding standard terminologies, we developed a semantic map on the basis of the knowledge graphs and the here introduced annotation helper plugin for Flyover. Flyover is a tool that converts structured data into resource description framework (RDF) triples and enables semantic interoperability. As a demonstration, we mapped data that are to be included in the STRONG AYA infrastructure. Results: The knowledge graphs provided a comprehensive overview of the large number of STRONG AYA concepts. The semantic terminology mapping and annotation helper allowed us to query data with incomprehensible terminologies, without changing them. Both the knowledge graphs and semantic map were made available on a Hugo webpage for increased transparency and understanding. Conclusion: The use of semantic web technologies, such as RDF and knowledge graphs, is a viable solution to overcome challenges regarding data interoperability and reusability for a federated AYA cancer data infrastructure without being bound to rigid standardized schemas. The linkage of semantically meaningful concepts to otherwise incomprehensible data elements demonstrates how by using these domain-agnostic technologies we made nonstandardized health care data interoperable.
Development of a core outcome set for adolescents and young adults with cancer
Anne-Sophie Darlington, Kirsty Way, Nicole Collaço, Charlotte Cairns, Simone Hanebaum, Silvie H. M. Janssen, Urška Košir, Martin McCabe, Daniel Stark, Samantha C. Sodergren, Winette T. A. van der Graaf, Olga Husson, STRONG AYA Consortium
The Lancet Oncology, 27(1), e45-e54 (2026). DOI
Adolescents and young adults aged 15–39 years diagnosed with cancer are faced with unique challenges, which affect key developmental milestones and can create complex care needs. The aim of this study was to reach international consensus on the minimum set of outcomes to measure through the development of a core outcome set (COS). The COS development followed published methodological standards. A literature review and interviews with adolescents and young adults with cancer and health-care professionals generated a comprehensive list of 129 outcomes, spanning clinical and patient-reported outcomes relevant to adolescents and young adults with cancer. A three-round online Delphi survey involving three stakeholder groups globally, was implemented to reach international consensus. Overall, 262 respondents participated in the Delphi survey and 126 (48·1%) completed three survey rounds. 59 outcomes met consensus in round 3 and were taken forward to the consensus meeting. The final COS consists of 20 outcomes, including two on-treatment specific domains and three off-treatment specific domains. This study developed an adolescents and young adult-specific COS that, when implemented in clinical care and research, will improve the relevance of research findings, enhance care delivery, and enable consistent data synthesis across studies. Future efforts will focus on refining measurement methods and ensuring global applicability.
Core Patient-Centered Outcomes for Adolescents and Young Adults with Cancer: A Comprehensive Review of the Literature from the STRONG-AYA Project
Silvie H. M. Janssen, Winette T. A. van der Graaf, Anna Hurley-Wallace, Carla Vlooswijk, Catarina S. Padilla, Charlotte Cairns, Connor J. Tyler, Emily I. Holthuis, Gudrun E. Rohde, et al
Cancer, 17(3), 454 (2025). DOI
This review provides an overview of relevant outcomes of AYAs with cancer in order to develop a core outcome set (COS: an agreed standardized collection of outcomes) for this population. A literature search was conducted, after which articles were reviewed by two independent researchers using Rayyan to screen articles by their title/abstract and their full text. The data were extracted using a tailored extraction framework by a team of reviewers. A total of 1631 of the 17,301 screened articles were included. Of the five core areas, functioning (47.0%) and epidemiology (44.9%) were covered most often, followed by physiological/clinical (42.4%), resource use (6.1%), and adverse events (4.5%). The most represented outcome domains include mortality/survival, delivery of care, outcomes relating to neoplasms, and emotional functioning/well-being. This literature review provides a foundation for the development of an AYA-specific COS to improve the relevance and efficiency of measuring outcomes, the pooling of (international) data, and the value of care for AYAs with cancer.
From Federated Learning to Publicly Facing Platform; Proof-of-Concept of the STRONG AYA Information Portal
Joshi Hogenboom, Aiara Lobo Gomes, Andre Dekker, Winette T. A. van der Graaf, Olga Husson, Leonard Wee
IOS Press 327, 591-592 (2025). DOI
Information on health data is often shrouded in exclusivity and sharing of such information is normally difficult and time-locked for numerous reasons. Federated learning (FL) regularly accelerates knowledge sharing, yet it is not fully exploited to alleviate overall data exclusivity and time constraints. Here, we present a proof-of-concept for a public FL-based info portal of the STRONG AYA Initiative.
Getting real about synthetic data ethics: Are AI ethics principles a good starting point for synthetic data ethics?
Danielle Shanley, Joshi Hogenboom, Flora Lysen, Leonard Wee, Aiara Lobo Gomes, Andre Dekker, Darian Meacham
EMBO reports 25, 2152-2155 (2024). DOI
The creation of synthetic data (SD) — ‘artificial’ data generated specifically with the goal of resembling ‘real-world’ data in order to replace or reduce the need for ‘real’ data — is booming. SD promises to be a viable alternative when data collection and data sharing may not be feasible or cost-effective. However, the increasingly prevalent use of SD for training and benchmarking in AI development is generating novel and pertinent ethical concerns that should be considered by developers and users of synthetic datasets and AI applications. This article provides an overview of issues arising from the rapid growth in SD generation and use to encourage further reflection and investigation within the research community.
Adolescent and young adult (AYA) patient involvement and engagement in European health care and research projects: expanding the scope of patient advocacy
Urska Košir, Flora Lysen, Nicola Unterecker, Tomaž Deželak, Erik Sturesson, Iryna Shakhnenko, Daniel Stark, Katie Rizvi, Anne-Sophie Darlington, Leonard Wee, Winette T. A. van der Graaf, Olga Husson
ESMO Open 10(3), 104478 (2025). DOI
Patient involvement and engagement (PI&E) in health care and research has gained prominence, shifting towards person-centred approaches and shared decision making. Patients actively participating in health care design and research lead to better quality and efficiency of care. However, implementing meaningful PI&E is challenging and requires adequate resources and evaluation frameworks so that it does not result in tokenism. This is particularly important when considering niche areas like adolescents and young adults (AYAs) with cancer. As AYAs’ unique needs continue to gain recognition, it is becoming increasingly important to incorporate their expertise and diverse perspectives in navigating care. Large-scale European consortia that focus specifically on AYAs offer opportunities to establish successful partnerships with AYAs in the design and creation of the next generation of equitable, diverse, and inclusive cancer care. Concrete actions for meaningful AYA PI&E are discussed.
Protocol for the development of a Core Outcome Set (COS) for Adolescents and Young Adults (AYAs) with cancer
Olga Husson, Silvie H. M. Janssen, Bryce B. Reeve, Samantha C. Sodergren, Christabel K. Cheung, Martin G. McCabe, John M. Salsman, Winette T. A. van der Graaf, Anne-Sophie Darlington
BMC Cancer, 24, 126 (2024). DOI
Background: Adolescents and young adults (AYAs) with cancer, defined as individuals aged 15–39 years at initial cancer diagnosis, form a unique population; they face age-specific issues as they transition to adulthood. This paper presents the protocol for the development of a core outcome set (COS) for AYAs with cancer. Methods: The methodological standards from the Core Outcome Measures in Effectiveness Trials (COMET) and the International Consortium for Health Outcomes Measurement (ICHOM) for COS development will guide the development of the COS for AYAs with cancer. The project will consist of the following phases: (1) define the scope of the COS; (2) establish the need for a COS in this field (3) assemble an international, multi-stakeholder working group; (4) develop a detailed protocol; (5) determine “what to measure” (i.e., outcomes); (6) determine “how to measure” (i.e., measures); and (7) determine “case-mix” variables. Conclusions: The development of a COS for AYAs with cancer will facilitate the implementation of efficient and relevant standards for data collection, both for clinical trials and in routine healthcare, thereby increasing the usefulness of these data to improve the value of the care given to these underserved young cancer patients.
Actionability of Synthetic Data in a Heterogeneous and Rare Health Care Demographic: Adolescents and Young Adults With Cancer
Joshi Hogenboom, Aiara Lobo Gomes, Andre Dekker, Winette T. A. van der Graaf, Olga Husson, Leonard Wee
JCO Clin Cancer Inform 8, e2400056 (2024). DOI
Research on rare diseases and atypical health care demographics is often slowed by high interparticipant heterogeneity and overall scarcity of data. Synthetic data (SD) have been proposed as means for data sharing, enlargement, and diversification, by artificially generating real phenomena while obscuring the real patient data. The utility of SD is actively scrutinized in health care research, but the role of sample size for actionability of SD is insufficiently explored. We aim to understand the interplay of actionability and sample size by generating SD sets of varying sizes from gradually diminishing amounts of real individuals’ data. We evaluate the actionability of SD in a highly heterogeneous and rare demographic: adolescents and young adults (AYAs) with cancer. Methods: A population-based cross-sectional cohort study of 3,735 AYAs was subsampled at random to produce 13 training data sets of varying sample sizes. We studied four distinct generator architectures built on the open-source Synthetic Data Vault library. Each architecture was used to generate SD of varying sizes on the basis of each aforementioned training subsets. SD actionability was assessed by comparing the resulting SD with their respective real data against three metrics—veracity, utility, and privacy concealment. Results: All examined generator architectures yielded actionable data when generating SD with sizes similar to the real data. Large SD sample size increased veracity but generally increased privacy risks. Using fewer training participants led to faster convergence in veracity, but partially exacerbated privacy concealment issues. Conclusion: SD is a potentially promising option for data sharing and data augmentation, yet sample size plays a significant role in its actionability. SD generation should go hand-in-hand with consistent scrutiny, and sample size should be carefully considered in this process.
Navigating Romantic Relationships and Sexual Intimacy: Challenges and Opportunities for Adolescents and Young Adults With Cancer – A Literature Review of Qualitative Studies
Nicole Collaço, Céline Bolliger, Charlotte Cairns, Samantha Sodergren, Olga Husson, Winette T. A. van der Graaf, Maria Rothmund-Grenier, Milou J. P. Reuvers, Gudrun E. Rohde, Anna L. Hurley-Wallace, et al
Psycho-Oncology, 34(9), e70285 (2025). DOI
This review explores the impact of cancer on adolescents’ and young adults’ (AYAs) romantic and sexual relationships, examining both challenges and opportunities. Qualitative studies on AYAs with cancer and/or their partners were searched based on a sub-set of studies identified in a previous literature review within the EU Horizon STRONG-AYA project, and supplemented with an additional search on romantic/sexual relationships. Articles were screened, reviewed in Rayyan and data extracted using a structured framework. Study quality was appraised, and findings were synthesised thematically. Twenty studies were included. Findings revealed how cancer altered AYAs’ sense of self, affecting the navigation of romantic relationships and sexual intimacy across four themes: (1) Romantic relationship development-connection, isolation and changing priorities: Disrupted romantic trajectories and fears of being undesirable hindered relationship formation, though some re-evaluated priorities, deepening emotional connections, (2) The disclosure dance: Finding the right time and way: AYAs struggled with when and how to disclose their diagnosis, with anxiety about burdening partners or altering dynamics. Open and honest communication fostered intimacy, (3) Body image, sexuality, intimacy, and fertility: Challenges in these areas impacted self-worth, but also led to sexual empowerment and adapted intimacy, (4) Partnerships and social support during cancer: Unsupportive partners heightened isolation, while supportive partners reinforced self-worth. Many AYAs experienced growth, resilience and a deeper understanding of themselves/their relationships. Results may inform age-appropriate interventions to adequately address the unique challenges AYAs with cancer face in navigating romantic relationships and sexual intimacy.
Decreased Heart Rate Variability Is Associated with Increased Fatigue Across Different Medical Populations: A Systematic Review
Sophie Maria Penfold, James Cunningham, Pauline Whelan, Martin G. McCabe, John Ainsworth
Pathophysiology, 32(3), 46 (2025). DOI
Background: Fatigue has been associated with poorer quality of life and increased morbidity in multiple clinical fields. Patients with autonomic dysfunction have been found to experience poorer physiological health, as well as having an increased risk of comorbidity and all-cause mortality. Heart rate variability (HRV) has been documented as a validated tool to assess autonomic function in clinical practice. The aim of this systematic review was to understand the relationship between fatigue and HRV in different medical populations. Methods: A systematic search was conducted in MEDLINE via Web of Science and Scopus. Results: A total of seventeen articles were identified for inclusion. Patients with Chronic Fatigue Syndrome were the most investigated population (n = 7), followed by cancer (n = 4) and Multiple Sclerosis (n = 4). The most implemented fatigue measure was the Multidimension Fatigue Inventory Scale used in four studies and HRV was monitored by electrocardiogram in nine studies. The most recorded and analysed domain for HRV was the frequency parameters. A significant association between increased subjective fatigue and imbalanced metrics of HRV (p < 0.05) was identified in fourteen articles. However, results from this review were heterogenous partly owing to the inconsistency with the instruments implemented to monitor HRV and measure fatigue. Additionally, only a small number of medical conditions were investigated, and the patients were predominately older adults (mean age 43.2) and women (64%). Conclusions: Despite these discrepancies, the reviewed evidence suggests that a rise in sympathetic activity and reduced parasympathetic tone are associated with an increased perception of fatigue in medical populations.
Minimum standards of specialist adolescent and young adult (AYA) cancer care units: recommendations and implementation roadmap
Urška Košir, Ana-Maria Totovina, Daniel Stark, Andrea Ferrari, Johan de Munter, Winette T. A. van der Graaf, Eveliene Manten-Horst, Katie Rizvi
ESMO Open, 10(10), 105829 (2025). DOI
In 2022, according to the Global Cancer Observatory, >115 000 adolescents and young adults (AYAs; aged 15-39 years) were diagnosed with cancer across Europe. Despite their unique biological and psychosocial needs, AYAs face disparity of access to specialised oncology services, particularly outside major urban centres in the east and south of Europe. Addressing these inequalities is essential to ensuring comprehensive and equitable cancer care. As part of EU4Health, the European Union Network of Youth Cancer Survivors (EU-CAYAS-NET) consortium was established to advance Europe’s Beating Cancer Plan. The project aimed to develop—with patient organisations at the helm—evidence-based recommendations to standardise and improve care across Europe, ultimately enhancing outcomes and quality of life for all young people.
Caught in the Middle: Identifying Gaps for Adolescents and Young Adults (AYAs) with Cancer – STRONG AYA Stakeholder Forum Report
Marion L’Hôte, Nora Lorenzo i Sunyer, Norbert Couespel
The STRONG-AYA Consortium held its first Stakeholder Forum ‘Caught in the Middle: Identifying Gaps for Adolescents and Young Adults with Cancer’ on 27 September 2023, bringing together 70 participants. The aim was to discuss the exclusive achievements of the project, provide a space for relevant stakeholders to provide feedback on the project’s ongoing activities and engage with the AYA Community on the project, beyond its current development and duration. This event report consequently focuses on the key messages and recommendations emanating from the Forum.