Top 10 research priorities for adolescent and young adult cancer in Canada: a James Lind Alliance priority setting partnership

Abstract

Objectives

Cancer in adolescents and young adults (AYAs; ages 15–39 years) is a rising global epidemic. Yet, AYAs remain an understudied population, and little is known about what research topics should be prioritised according to those with lived experience. The AYA Cancer Priority Setting Partnership (PSP) was established to identify the top 10 research priorities for AYA cancer in Canada according to patients, caregivers, and clinicians.

Design

This project followed the James Lind Alliance (JLA) Priority Setting Partnership (PSP) methodology that included two national cross-sectional surveys and a final priority setting workshop following an adapted nominal group technique.

Setting

A national sample was recruited to participate from across Canada.

Participants

Participants were patients, caregivers, and clinicians with lived personal and/or professional experience of AYA cancer in Canada.

Outcome measures

In the first survey, open-ended responses were collected from participants about questions they would like answered by research. Responses were collated into overarching summary questions and a literature search was undertaken to verify if questions were true uncertainties and not fully answered by existing evidence. Unanswered questions were ranked by participants in a second survey. The top-ranked questions were prioritised through consensus at the final priority setting workshop. The final outcome was the top 10 priorities for AYA cancer research in Canada.

Results

In the initial survey, 1916 potential research questions were submitted by 275 patients, caregivers, and clinicians. Following data processing, summary question formation, and the evidence check, 58 questions were put forward for interim prioritisation in a second survey (n=285 patients, caregivers, and clinicians). The top 20 questions from the interim prioritisation were ranked at the final priority setting workshop attended by a diverse group of 23 patients, caregivers and clinicians from across Canada. The resulting top 10 priorities reflect topics across the cancer continuum including: diagnostic delays, screening and early detection, novel therapies, psychosocial impacts, end-of-life concerns, and survivorship issues.

Conclusions

This patient-directed research agenda will guide researchers, funding agencies, and policymakers to ensure that future research is aligned with what matters most to the AYA cancer community.

STRENGTHS AND LIMITATIONS OF THIS STUDY.

• The Adolescent and Young Adult (AYA) Cancer Priority Setting Partnership (PSP) used the rigorous James Lind Alliance PSP methodology to establish the top 10 research priorities for AYA cancer in Canada according to individuals with lived experience and those who care for them.

• The AYA Cancer PSP was conducted in partnership with a network of over 30 community organisations to ensure that the top 10 priorities generated from this project were inclusive and reflective of the diversity of the broader community’s lived experiences.

• The study surveys were available in both Canadian official languages (ie., English and French) and individuals from nearly all of Canada’s provinces and territories participated.

• While we captured a wide range of perspectives throughout the project, some voices (e.g., younger AYAs, certain racial groups) were under-represented.

Introduction

Cancer in adolescents and young adults (AYAs; ages 15–39 years¹,²) is a rising global epidemic.³ Over the last 20 years, the overall incidence of cancer increased significantly in AYAs despite decreases in the adult population.¹ This trend is alarming given the number of formative years of life impacted for AYAs. Indeed, there is increasing recognition that AYAs are an oncology population distinct from other cohorts. The most common types of cancer diagnosed in this age group are different from those diagnosed in children and older adults⁴ with unique biological features requiring specialised treatment considerations.⁵ AYAs are also more likely than other groups to experience delays in diagnosis.^{6 7} While most AYAs diagnosed with cancer in high-income countries are expected to survive, improvements in survival rates have not kept pace with those seen in other age groups, due in part to these challenges.⁸ Further, the AYA developmental period is marked by the emergence of independence, identity development, and exploration of romantic relationships, family planning, and careers.⁹ Cancer and the long-term effects of treatment threaten the attainment of these critical life milestones. Most AYAs will live 50–60 years beyond their diagnosis¹ and face enduring physical (e.g., infertility, disability) and psychosocial (e.g., financial toxicity, mental health difficulties, disruptions to education and employment) sequelae that directly impact daily functioning, quality of life, and productivity.^{10 11} Despite the significant personal and societal impacts of AYA cancer, this population remains significantly understudied.^{1 12} Rigorously conducted and clinically actionable research across the cancer continuum is urgently needed to improve outcomes for AYA patients.

While national and international bodies have made systems-level recommendations for AYA cancer care research and practice,113,15 little is known about the topics that should be prioritised for research according to those with lived experience. Indeed, the research priorities of the scientific community do not necessarily align with those with lived experience.¹⁶ For example, scientists and industry prioritise pharmacological trials two to five times more frequently than patients, caregivers and clinicians.¹⁷ This mismatch in priorities is one of the key contributors to research waste¹⁸ and reasons for limited impact of research on patient outcomes.¹⁹ Studies have demonstrated that the equal engagement of individuals with lived experience in establishing research priorities is associated with improved research relevance, quality, and influence on practice and policy.^{20 21}

The James Lind Alliance (JLA) has established a systematic process for identifying the key questions that patients, caregivers and clinicians want answered by research ultimately resulting in a ‘top 10’ list of research priorities.²² It is the gold standard approach for establishing patient-oriented research agendas and has been applied to over 150 clinical conditions,²³ including cancer in children^{24 25} and young people (<24 years of age).²⁶ Beyond establishing research priorities, a number of JLA priority setting projects have resulted in the development of new strategic partnerships and financial investments targeted specifically at advancing the research areas prioritised.²⁷ However, no patient-oriented research agenda focused on the AYA population (e.g., 15–39 years) currently exists. Establishing a patient-identified, caregiver-identified and clinician-identified research agenda for AYA cancer is necessary to drive improvements in care and outcomes for this population. Thus, the aim of the ‘AYA Cancer Priority Setting Partnership’ project was to conduct a JLA research prioritisation study to establish the top 10 research priorities for AYA cancer in Canada.

Methods

Study design

The study followed the established JLA Priority Setting Partnership (PSP) process²² and the protocol was previously published.²⁸ The JLA PSP process is a method of research prioritisation that follows four integral principles: (1) equal involvement; (2) inclusivity; (3) transparency; and (4) a commitment to using and contributing to the evidence base.²² While the method is flexible and responsive, the JLA process generally follows a standard sequence of events including: (1) establishing the team; (2) gathering initial questions; (3) summarising questions and checking the evidence to ensure they are unanswered; (4) prioritising a short-list of questions; and (5) reaching consensus on the top 10 priorities in a final priority setting workshop. Participants provided informed consent for each phase of the project by checking a box at the end of a corresponding online consent form to indicate their agreement to participate. The study followed the Reporting Guideline for Health Research Priority Setting with Stakeholders (see online supplemental materials).²⁹

Step 1: Establishing the team

The AYA Cancer PSP was co-led by two clinician-scientists in AYA cancer (PRT and FSMS) and an AYA cancer patient advocate (CT). With the co-leads, the project was governed by an expert Steering Group that oversaw all activities (e.g., establishing the scope, approving all study materials, assisting with data collection, analysis and summary question formation, and providing expert guidance regarding evidence checking) and chaired by a neutral JLA Adviser (TRad). The Steering Group was comprised of 16 members including patients (n=7), bereaved caregivers (n=2) and clinicians (n=7). Steering Group members were selected through a national call for expressions of interest (N=78 responses received) and represented a broad range of experiences. Patient members of the Steering Group included AYAs currently undergoing treatment as well as those who had completed treatment, while clinician members represented a range of disciplines, including paediatric oncology, medical oncology and palliative care, nursing, psychology, social work and rehabilitation. Further information on the selection of the Steering Group is described elsewhere.²⁸ The Steering Group met monthly between April 2023 and January 2025. Consistent with best practices in patient engagement in research,¹⁹ patient and caregiver members were compensated for their time and expertise. In addition to the Steering Group, a network of over 30 non-profit organisations (see box 1), who agreed to formally partner on the project and disseminate calls for expressions of interest, survey recruitment, and information on the final priorities to their communities, was established. AYA Can—Canadian Cancer Advocacy (a national peer-led advocacy organisation aimed at improving the experiences and outcomes for Canadian AYAs affected by cancer)³⁰ was the primary partnering community organisation. The project was also supported by a research team with relevant content and methodological expertise (JLB, KB, SB, SNG, LJ and MN).

Box 1. AYA Cancer PSP Community Partners.

• Alberta Strategy for Patient-Oriented Research SUPPORT Unit

• AYA CAN - Canadian Cancer Advocacy

• Brain Tumour Foundation of Canada

• Canadian Cancer Society

• Canadian Partnership Against Cancer

• Canadian Cancer Survivor Network

• Canadian Virtual Hospice

• Canadian Breast Cancer Network

• Canadian Cancer Clinical Trials Network

• Canadian Centre for Applied Research in Cancer Control

• Canadian Cancer Research Alliance

• Canadian Association of Nurses in Oncology

• Canadian Association of Psychosocial Oncology

• Colorectal Cancer Resource & Action Network

• C17 Council - Children’s Cancer & Blood Disorders

• Lung Cancer Canada

• Leukemia & Lymphoma Society

• Melanoma Canada

• Oneball Charitable Cancer Organization

• On the Tip of the Toes Foundation

• Pink Pearl Canada

• Queering Cancer

• Quebec Cancer Coalition

• Rethink Breast Cancer

• Save Your Skin Foundation

• Sarcoma Cancer Foundation of Canada

• Team Shan – Breast Cancer Awareness for Young Women

• The Pink Wig Foundation

• Young Adult Cancer Canada

Scope

This PSP sought to identify research priorities from AYAs living with and beyond cancer in Canada, caregivers of AYAs in Canada, and clinicians who provide direct care to AYAs in Canada. We used the Canadian Partnership Against Cancer definition of adolescence and young adulthood, which includes those diagnosed with cancer between the ages of 15–39 years.^{1 2} The scope was intentionally broad and invited all possible research questions pertaining to diagnosis, treatment, survivorship and end-of-life care for AYAs with cancer. According to the JLA, possible questions for research are considered out-of-scope if they did not relate to the project topic (i.e., AYA cancer) and/or if they are not researchable (i.e., single word responses, requests for information or advice or issues around awareness).²²

Step 2: Gathering initial questions for research (Survey #1)

An initial online survey was developed by the Steering Group to collect research questions that the AYA cancer community wanted to see answered by research. The survey was developed following established JLA methodology and was informed by surveys used in past JLA PSPs.^{31 32} Individuals were eligible to take part if they identified as AYAs living with and beyond cancer in Canada (diagnosed between the ages of 15–39 years of age), caregivers of AYAs in Canada, and clinicians who provide direct care to AYAs in Canada, in accordance with the scope of the PSP specified in Section 2.2.1. Participants were asked to provide open-ended responses about what questions they would like answered related to AYA cancer in the following areas: screening and/or diagnosis, treatment and/or care, living beyond cancer, and end-of-life care. Participants were also invited to add responses in areas beyond those specifically outlined and reported on their demographic characteristics. See online supplemental materials for the survey questions. The survey was piloted by members of the Steering Group prior to the start of data collection to optimise clarity, usability, and technical functionality and minor revisions were made accordingly. To reduce barriers to participation and mitigate potential non-response bias, the survey was made available in both English and French. It was disseminated using a multichannel sampling approach to maximise reach across relevant groups. Distribution occurred through a network of more than 30 national community organisations, including organisations serving under-represented cancer populations (e.g., Queering Cancer) as well as through the Steering Group, organisational mailing lists, and social media. Recruitment efforts were monitored during the data collection period to ensure balanced participation, and dissemination strategies were adjusted as needed to support inclusivity across stakeholder groups. Data were collected using Qualtrics survey software (Qualtrics, Provo, Utah, USA) from September to December 2023. In accordance with best practices for survey-based research,³³ several precautions were taken to prevent fraudulent participation including: enabling bot detection technology, reviewing submissions for duplicate responses and reviewing submissions for inconsistencies in responses. No compensation was provided to survey participants.

Step 3: Processing initial Questions, forming summary questions, checking the evidence

Responses collected from Survey #1 (Step 2) were processed by PRT and TRan with regular discussion and review by the Steering Group. Each response was given a reference number to maintain an audit trail. Responses submitted in French were translated to English by a professional translator prior to analysis. After removal of the responses deemed out-of-scope, submissions were coded into broad categories and summarised into overarching summary questions. Steering Group members were paired (patient/caregiver and clinician) and assigned batches of questions to review to ensure they accurately reflected the raw data. Changes to the summary question wording were made based on their feedback. Then, in line with JLA guidelines,²² a focused literature search was conducted on 18 July 2024 using Trip Database Pro³⁴ for recent and relevant secondary evidence (e.g., reviews, clinical guidelines and position statements) published within the last 5 years (i.e., 2019–2024) to verify whether each question was a true uncertainty and not fully answered by existing evidence. Trip Database searches more than 75 sources of evidence including international guidelines, PubMed and PubMed Central, Cochrane Database, Database of Abstracts of Reviews of Effects, and core medical sources including the BMJ, Lancet, JAMA and NEJM. A comprehensive set of search terms was established based on past literature.³⁵ See online supplemental materials for the full search and selection strategy. Resulting titles and abstracts and full text articles were screened by two reviewers (PRT and TRan) using Covidence review management software (Veritas Health Innovation, Melbourne, Australia) and checked against each summary question. Consistent with past PSPs,³⁶ summary questions were considered unanswered if there was no review or guideline on the topic, if existing reviews or guidelines concluded there was insufficient evidence in an area, or if the evidence outlined was overly narrow (ie, lack of research, focus on a specific disease population only). The final question verification form was reviewed and approved by the Steering Group.

Step 4: Interim priority setting (Survey #2)

The second survey (available in English and French) was administered online from September to November 2024 using Qualtrics survey software (Qualtrics, Provo, Utah, USA). The survey was open to AYAs diagnosed with cancer, caregivers, and clinicians in Canada and distributed through the same channels as Step 2. Participants were asked to identify their top 10 priorities from the list of 58 summary questions presented in random order and to rank the selected questions from most to least important. Demographic information was also collected (see online supplemental materials for the survey questions). The 58 summary questions were assigned weighted scores according to their rank order, with higher-ranked questions receiving more points. Total scores for each question were calculated separately for each respondent group (patients/caregivers and clinicians). To ensure equal consideration of priorities across groups, the 20 highest-ranked questions—representing an equal number of the top ranked questions for patients/caregivers and clinicians—were identified as the shortlist of questions to advance to Step #5. The same precautions were taken as with Survey #1 and respondents were not compensated for their participation.

Step 5: Final workshop

The final workshop was held virtually using Zoom (Zoom Communications, San Jose, California, USA) over two half-days (16–17 November 2024). A total of 24 patients, caregivers, and clinicians from across Canada were recruited to participate via an open call for expressions of interest. Participants were selected purposively by the co-leads from the pool of those who expressed interest (N=59) to enhance the diversity of voices based on geography, gender, diagnosis, and perspective. Prior to the workshop, participants were provided with preparatory materials, including the list of 20 shortlisted questions, and were asked to identify their top and bottom three questions. The workshop was chaired by the project JLA Adviser (TRad) and employed an adapted nominal group technique, incorporating both small-group and large-group discussions to achieve consensus on the top 10 priorities.²² On the first day, participants received an overview of the project and workshop objectives before being assigned to three small groups comprising a mix of patients/caregivers and clinicians. Each group was facilitated by a trained JLA Adviser to ensure balanced participation. Discussions began with participants sharing their top and bottom three priorities, followed by a preliminary ranking of the 20 shortlisted questions. At the end of day 1, rankings from each group were averaged and presented back to participants in a large group discussion. On the second day, participants were reallocated to new small groups, where JLA Advisers facilitated further discussion to refine the rankings established on day 1. Rankings were again averaged across groups, resulting in the final top 10 list of priorities, which was presented to the large group prior to the end of the workshop. Participants were compensated with a $C100 electronic gift card for taking part.

Patient and public involvement

Patient and public involvement was at the core of this project, which was co-built together with people with lived experience from the very start. The project was co-led by two clinician scientists (PRT and FSMS) and a patient advocate (CT). The co-leads made all plans and decisions collaboratively, including the establishment of the Steering Group. Patients and caregivers were engaged as equal members of the Steering Group in all stages of the project from protocol development to data collection and analysis, and knowledge dissemination (e.g., co-presenters on presentations, co-authors on publications including this article) and were compensated for their time and expertise.

Results

Gathering initial questions for research (Survey #1)

In the first survey, 275 participants provided 1916 possible questions for research, including four individuals who submitted 33 questions in French. Only one record was flagged as a potential fraudulent participant due to inconsistent responding and thus this participant was excluded. Of the 275 valid participants, 161 (58%) identified as a patient, 92 (33%) identified as clinicians, and 35 (13%) identified as a caregiver. Participants came from 11 provinces and territories (85%) and 55 (20%) identified with a racialised background. Patients had histories of a range of cancer diagnoses, and most had completed treatment (40%) or were on active surveillance (15%). Of the caregivers, the majority identified as parents (37%) or partners (20%) and about one-third (34%) were bereaved. Clinicians represented a range of healthcare professionals, most commonly physicians (n=36; 39%). See table 1 for complete demographics.

Table 1. Participant demographic characteristics.

Characteristic	No. (%)
	Survey 1 (N=275)	Survey 2 (N=285)	Final workshop (N=23)
Role*
Person diagnosed with cancer as an AYA	161 (58.6)	146 (51.2)	10 (43.5)
Family, friend, or loved one	35 (12.7)	69 (24.2)	6 (26.1)
Clinician	92 (33.5)	94 (33.0)	8 (34.8)
Gender identity*
Cisgender woman	212 (77.1)	221 (77.5)	15 (65.2)
Cisgender man	46 (16.7)	49 (17.2)	8 (34.8)
Genderqueer	5 (1.8)	3 (1.1)	0
Gender non-conforming	5 (1.8)	1 (0.4)	0
Non-binary	4 (1.5)	4 (1.4)	0
Trans man	1 (0.4)	1 (0.4)	0
Trans	1 (0.4)	1 (0.4)	0
Other	1 (0.4)	1 (0.4)	0
Prefer not to answer	9 (3.3)	9 (3.2)	0
Sexual orientation*
Heterosexual/straight	232 (84.4)	238 (83.5)	23 (100.0)
Bisexual	16 (5.8)	21 (7.4)	0
Queer	15 (5.5)	16 (5.6)	0
Pansexual	14 (5.1)	8 (2.8)	0
Asexual	8 (2.9)	3 (1.1)	1 (4.4)
Gay/Lesbian	2 (0.7)	6 (2.1)	0
Two-spirit	0	1 (0.4)	0
Other	1 (0.4)	0	0
Prefer not to answer	7 (2.5)	10 (3.5)	0
Race/Ethnicity*
White	237 (86.2)	232 (81.4)	18 (78.3)
South Asian	13 (4.7)	16 (5.6)	3 (13.0)
East Asian	12 (4.4)	15 (5.3)	1 (4.4)
Indigenous	8 (2.9)	10 (3.5)	0
Middle Eastern	7 (2.6)	6 (2.1)	1 (4.4)
Black	3 (1.1)	4 (1.4)	0
Latin American	4 (1.5)	3 (1.1)	0
Southeast Asian	3 (1.1)	3 (1.1)	0
Prefer to self-identify	5 (1.8)	3 (1.1)	0
Prefer not to answer	5 (1.8)	11 (3.9)	0
Province or territory of primary residence
Ontario	99 (3.0)	108 (37.9)	8 (34.8)
Alberta	65 (23.6)	78 (27.4)	3 (13.0)
British Columbia	35 (12.7)	54 (19.0)	2 (8.7)
Manitoba	28 (10.2)	11 (3.9)	4 (17.4)
Quebec	18 (6.6)	13 (4.6)	3 (13.0)
Newfoundland and Labrador	9 (3.3)	8 (2.8)	1 (4.4)
Nova Scotia	7 (2.6)	5 (1.8)	2 (8.7)
Saskatchewan	6 (2.2)	5 (1.8)	0
New Brunswick	4 (1.5)	1 (0.4)	0
Northwest Territories	3 (1.1)	0	0
Yukon	1 (0.4)	1 (0.4)	0
Prince Edward Island	0	1 (0.4)	0
Age at diagnosis†
15–19 years	15 (9.3)	20 (13.7)	2 (20.0)
20–29 years	46 (28.6)	41 (28.1)	5 (50.0)
30–39 years	61 (37.9)	58 (39.7)	3 (30.0)
Primary cancer diagnosis†
Breast	26 (16.2)	32 (21.9)	1 (10.0)
Lymphoma	26 (16.2)	30 (20.6)	1 (10.0)
Brain/CNS	12 (7.5)	8 (5.5)	1 (10.0)
Sarcoma	11 (6.8)	10 (6.9)	0
Colorectal	11 (6.8)	8 (5.5)	0
Leukaemia	7 (4.4)	6 (4.1)	0
Cervical	5 (3.1)	4 (2.7)	0
Testicular	4 (2.5)	3 (2.1)	1 (10.0)
Thyroid	4 (2.5)	3 (2.1)	1 (10.0)
Melanoma	1 (0.6)	2 (1.4)	1 (10.0)
Other	16 (9.9)	14 (9.6)	4 (40.0)
Current treatment status†
Currently receiving treatment	34 (21.1)	37 (25.3)	2 (20.0)
Completed treatment	65 (40.4)	51 (34.9)	6 (60.0)
On active surveillance	24 (14.9)	32 (21.9)	2 (20.0)
Healthcare profession‡
Physician	36 (39.1)	32 (34.0)	3 (37.5)
Nurse	17 (18.5)	17 (18.1)	0
Psychologist	6 (6.5)	9 (9.6)	1 (12.5)
Social worker	5 (5.4)	8 (8.5)	1 (12.5)
Physiotherapist	4 (4.4)	4 (4.3)	1 (12.5)
Dietician	1 (1.1)	1 (1.1)	0
Pharmacist	1 (1.1)	1 (1.1)	0
Occupational therapist	1 (1.1)	0	0
Other	10 (10.9)	6 (6.4)	2 (25.0)

^*Respondents could select more than one response, responses may sum to greater than the total number of respondents.

^†Includes respondents who identified as patients: n=161 for survey 1, n=146 for survey 2, n=10 for the final workshop.

^‡Includes respondents who identified as a clinician: n=92 for survey 1; n=94 for survey 2; n=8 for the final workshop. AYA, adolescents and young adults.

CNS, central nervous system.

Processing initial questions, forming summary questions, checking the evidence

Of the 1916 initial questions submitted in the first survey, 1559 (81%) were deemed to be in-scope and were aggregated into 58 summary questions. For the evidence check, 5220 titles and abstracts and 274 full text articles were screened. A total of 207 articles (including 192 reviews and 15 guidelines) met the inclusion criteria and were reviewed against the summary questions (figure 1; see online supplemental materials for full list of reviewed articles). Following the evidence review, the Steering Group agreed that no questions were adequately answered by the evidence. Thus, all 58 summary questions were retained for the second survey (see online supplemental materials).

Figure 1. Flow diagram of responses from Survey #1 to the final workshop.

Interim priority setting (Survey #2)

A total of 285 individuals completed the interim priority setting survey, including five in French. No respondents were flagged as potentially fraudulent. Of those who participated, 146 (51%) identified as an AYA patient, 94 (33%) were clinicians and 69 (24%) identified as a caregiver (table 1). The 10 highest-ranked summary questions for the two respondent groups (patients/caregivers and clinicians) were examined separately (see table 2). As there was an overlap in the 10 highest ranked questions across the two groups, the next highest ranked questions from each group were selected until a list of 20 questions representing an equal balance of questions derived from the two groups was generated. The top 20 list of questions was put forward to the final prioritisation workshop.

Table 2. Rankings of the top 20 questions in Survey #2 (N=285).

Combined rank in survey 2	Summary question	Patient/ caregiver rank	Clinician
1	What are the impacts of a cancer diagnosis on the mental health of AYAs and how can their mental health be supported across the cancer continuum?	1	8
2	What are the physical and psychosocial long-term and late effects of cancer treatment for AYAs and how can AYAs be best monitored and supported for these long-term and late effects?	3	1
3	What challenges do AYAs face with transitioning off treatment and living beyond cancer (eg, return to work/school, reconnecting with peers, rehabilitation) and how can they be better supported with these challenges?	4	3
4	How can we improve access to healthcare services for AYAs (primary care, screening, psychosocial support, palliative care, follow-up care, rehabilitation, etc) before, during and after cancer treatment?	5	5
5	What are the most effective methods to monitor for cancer recurrence and secondary cancers in AYAs?	2	32
6	Why do many AYAs experience delays in cancer diagnosis and how can the diagnostic process be improved?	6	16
7	How can we improve screening for and early detection of cancer in AYAs in the general population and what are the outcomes associated with more inclusive screening practices?	7	15
8	What are the current gaps in care for AYAs across the cancer continuum according to patients and families and what services would they find the most beneficial?	15	2
9	How can we improve the development and implementation of novel treatments and testing protocols (eg, genomic testing) to improve outcomes for AYA cancers, including for relapse and rare cancers?	12	11
10	How can fertility and family planning experiences and outcomes be improved for AYAs across the cancer continuum?	14	6
11	What is the role of allied health (eg, physiotherapy, occupational therapy, psychology, social work, art therapy) in supporting AYAs and how can these services be made part of standard care across the cancer continuum?	8	12
12	Why are rates of some cancers increasing in AYAs and how can these increases be addressed?	13	56
13	What is the experience and impact of fear of cancer recurrence for AYAs and how can these fears be managed?	11	24
14	What are the impacts of cancer on the quality of life of AYAs and how can they be supported to live well both with and beyond cancer?	9	25
15	What factors lead to an increased risk of cancer among AYAs, and what preventative measures can help to reduce the risk?	17	18
16	What are the financial impacts of cancer on AYAs over their lifetime and how can these impacts be mitigated?	10	34
17	What are the post-treatment needs of AYAs living with or beyond cancer and how can follow-up services better meet their needs?	20	9
18	What are the end-of-life care needs and preferences of AYAs with cancer and their families, and what are the best ways to provide support for AYAs and families facing end-of-life?	36	4
19	How long does it take for AYAs to receive a cancer diagnosis and treatment from initial symptom onset?	16	46
20	What are the psychological, social and financial impacts on the friends and family of AYA patients with cancer and survivors and how can they be better supported across the cancer continuum.	18	27

AYA, adolescent and young adult.

Final workshop

24 participants were selected based on diversity (geographic, gender, diagnosis, or perspective) to participate in the final workshop. One participant was unable to attend on the day of the workshop due to illness, thus the final workshop was ultimately attended by 23 individuals. Participants included 10 (43%) patients, 8 (35%) clinicians, and 6 caregivers (26%; n=4 bereaved). See table 1 for full demographic characteristics. Across the three small groups (n=7–8 participants per group), participants described early difficulties with ranking the shortlisted questions, as each was perceived as a meaningful priority. However, following two rounds of group-based prioritisation, participants reached consensus on the top 10 priorities for AYA cancer (table 3). There was a high degree of agreement in priorities across the groups; all three groups ranked the top two priorities identically. Moreover, two groups had nine and one group had eight of their top 10 priorities represented in the final top 10 list (see online supplemental materials for all group rankings).

Table 3. The top 10 priorities for AYA cancer research.

Rank	Question
1	Why do many AYAs experience delays in cancer diagnosis and how can the diagnostic process be improved?
2	How can we improve access to healthcare services for AYAs (primary care, screening, psychosocial support, palliative care, follow-up care, rehabilitation, etc) before, during and after cancer treatment?
3	How can we improve the development and implementation of novel treatments and testing protocols (eg, genomic testing) to improve outcomes for AYA cancers, including for relapse and rare cancers?
4	How can fertility and family planning experiences and outcomes be improved for AYAs across the cancer continuum?
5	What are the end-of-life care needs and preferences of AYAs with cancer and their families, and what are the best ways to provide support for AYAs and families facing end-of-life?
6	What are the impacts of a cancer diagnosis on the mental health of AYAs and how can their mental health be supported across the cancer continuum?
7	What challenges do AYAs face with transitioning off treatment and living beyond cancer (eg, return to work/school, reconnecting with peers, rehabilitation) and how can they be better supported with these challenges?
8	What are the most effective methods to monitor for cancer recurrence and secondary cancers in AYAs?
9	What are the financial impacts of cancer on AYAs over their lifetime and how can these impacts be mitigated?
10	How can we improve screening for and early detection of cancer in AYAs in the general population and what are the outcomes associated with more inclusive screening practices?

AYA, adolescent and young adult.

Conclusions

Using the JLA PSP methodology, this project brought together diverse AYAs, their caregivers, and clinicians in Canada to establish the top 10 most important priorities for AYA cancer research. The resulting top 10 questions reflect topics that span the phases of the cancer continuum (e.g., prevention, screening, diagnosis, treatment, survivorship, end-of-life) and pillars of health research (e.g., biomedical, clinical, health services, and social, cultural, environmental, and population health research).³⁷ During the evidence check step of the project, it was determined that none of the 58 summary questions had been fully answered by past research, highlighting the significantly understudied nature of patient-oriented research topics in AYA cancer. The top 10 priorities identified in this project chart a path forward for research teams and funding agencies to advance research that will improve outcomes that matter most to people with lived experience.

Over the past decade, several cancer-related JLA PSPs have been conducted, including on childhood cancer (individuals diagnosed <18 years)^{24 38} and teenage and young adult cancer (individuals diagnosed between 13 and 24 years).²⁶ The current study is the first to establish a patient-oriented research agenda focused exclusively on the entire AYA developmental trajectory (eg, 15–39 years), as defined by the Canadian Partnership Against Cancer¹ and National Institutes of Health.² While some overlap exists in topics prioritised across various cancer-related PSPs (e.g., survivorship, relapse, psychosocial issues),2426 39,45 this project highlights the unique priorities of those diagnosed with cancer as AYAs that have not been previously captured, such as the financial impacts of cancer and family planning concerns. In recent years, there has been increasing attention given to the issue of oncofertility,46,48 yet this care is still not universally accessible to patients^{49 50} and, interestingly, it was not the top ranked priority of importance, perhaps due to the attention it has already received. There is also far less understood about other family planning options (e.g., adoption, surrogacy) for young people diagnosed with cancer.

The top-ranked priority identified in this project focused on the issue of diagnostic delays experienced by AYAs with cancer. Compared with children and adults diagnosed with cancer, AYAs are more likely to face significantly longer intervals between symptom onset and diagnosis.^{6 7} While findings on the relationship between diagnostic delays and clinical outcomes (e.g., survival, stage at diagnosis) are mixed,^{51 52} actual or perceived delays are associated with poorer patient-reported outcomes such as greater psychological distress, worse quality of life, and impaired trust in providers and the healthcare system.^{53 54} The reasons for diagnostic delays are complex (e.g., limited patient and clinician knowledge, lack of screening programmes for AYAs, low provider suspicion of cancer in young people, and prolonged wait times due to system capacity)⁵⁵ and align with several other top ranked priorities in this PSP (e.g., access to care, screening and early detection) and others.²⁶ Addressing the issue of delayed diagnosis has the potential for significant downstream improvements for the care and outcomes of AYAs.

National and international bodies have outlined priorities to guide future research in AYA cancer.^{13 15} Such initiatives, with limited involvement of people with lived experience, have resulted in research agendas focused heavily on biological, epidemiological, and health systems-related topics. The priorities established through this project address topics across the spectrum of health research, with many questions necessitating transdisciplinary approaches to result in meaningful change. Indeed, the need for research on the development of novel treatments and testing protocols tailored to AYA cancers was identified as a top priority. Yet, about half of the top 10 priorities focus on topics that include the psychological and social impacts of cancer on AYAs. Psychosocial issues are a primary concern for AYA patients across the cancer continuum and are among the greatest unmet need,^{56 57} yet receive only a small minority of cancer research funding.¹ Findings from this project highlight the need for further investments in research on the psychosocial dimensions of AYA cancer.

Beyond the top 10 priorities, this project identified several additional topics for research deemed important to the AYA cancer community. Topics ranked in positions 11–20 represent additional priority areas for research discussed at the final workshop, such as focused work on fear of cancer recurrence in AYAs, transitions from paediatric to adult cancer care, and the need for standardised AYA cancer care (see table 2). While not ranked in the top 10, all priorities identified through this project were identified as important to the community and warrant further investigation.

This study had numerous strengths, including the use of the JLA PSP methodology—the gold standard approach for establishing patient-oriented research agendas.²² We were guided by a trained JLA Advisor who ensured adherence to the methodology and engaged a network of over 30 organisations to support recruitment and dissemination. This ultimately resulted in the engagement of participants across study activities that represented diverse perspectives. The study surveys were available in both Canadian official languages (i.e., English and French), and across the stages of the project, we recruited individuals residing in 12/13 (92%) of Canada’s provinces and territories.

Some limitations must also be acknowledged. First, the majority of AYA patients who participated were diagnosed at the age of 20 or older. While the vast majority (90%+) of cancer diagnoses in individuals between the ages of 15–39 years occur in individuals over the age of 20,⁴ the current study may not have fully captured the needs of those diagnosed between the ages of 15–19 years, which may be better described in past PSPs focused on the needs of younger patients with cancer.^{24 26 38} Second, while approximately 20% of participants in the study identified with a racialised background (compared with approximately 30% of the Canadian general population),⁵⁸ there is under-representation from some racial backgrounds (e.g., individuals who self-identify as Indigenous and Black) who may have distinct needs and priorities.⁵⁹ Finally, this study focused on the perspectives of people with lived experience in Canada, and while many of the prioritised topics are likely generalisable to the AYA cancer experience, those living in other countries may have unique experiences and priorities, particularly as they relate to healthcare systems and services.

In sum, the AYA Cancer PSP identified the top research priorities for AYA cancer according to patients, caregivers, and clinicians. This patient-oriented research agenda is primed for action by researchers, funding agencies, patient organisations, and policymakers to ensure that future AYA cancer research is in line with what matters most to patients, caregivers, and clinicians to improve outcomes and care for AYA patients and their families.

Data availability statement

Data are available upon reasonable request.