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Abstract

Introduction

The purpose of this study was to identify patterns and themes that support participant engagement in patient-partnered cancer genomics research.

Methods

The Osteosarcoma (OS) and Leiomyosarcoma (LMS) Projects of Count Me In allow any patient with OS and LMS in the US and Canada to contribute their health information, tumor samples, and lived experience to an aggregated, public research database. We conducted in-depth interviews with research partners, including patients, caregivers, and advocates, who were purposefully sampled to ensure inclusion of racial and ethnic minorities, those with less than college education, and adolescents (age 12-17). Coding and analysis were conducted by the research team using NVivo to identify themes that support engagement.

Results

Ten patients, ten caregivers, and six advocates were interviewed. Seven themes were identified that support participant engagement: (a) motivation, (b) respect, (c) trust, (d) inclusivity, (e) relationship, (f) engagement, and (g) empowerment. Research partners were motivated to serve others, play a part in scientific discovery, and play a role in a novel initiative. Respect was supported through timeliness in communication or follow-up, an appropriate amount of time and information requested, and an acknowledgement that illness may prevent participation. Trust was developed through ensuring adequate privacy/confidentiality safeguards and demonstrating transparency. Inclusivity was demonstrated through showcasing broad representation and mitigating technical barriers. Research partners wanted to feel a relationship with, and engaged and empowered by, researchers. Adolescents reported their parents were more engaged than they were.

Conclusions

Research partners, including patients, caregivers, and advocates, have a strong desire to engage with researchers. We identified seven themes to support engagement. Researchers can optimize their communication and operations to support participant engagement in cancer genomics research.

Plain Language Summary

How can research teams support engagement in patient-partnered cancer genomics research? Through semi-structured interviews with patients, caregivers, and advocates, we identified seven themes that supported participant engagement: (a) motivation to help others or advance scientific knowledge, (b) feeling respected through the process, (c) trust in the research team, (d) inclusivity, (e) experiencing a bidirectional relationship with the research team, (f) feeling engaged as a meaningful partner in the research, and (g) empowerment. Researchers can support participant engagement by proactive communication and operational efforts that support these themes.

Keywords

bone sarcoma soft tissue sarcoma adolescent and young adult cancer genomics research

Introduction

Osteosarcoma (OS) and leiomyosarcoma (LMS) are rare cancers arising from the bone and smooth muscle, respectively, with fewer than 2000 new OS and LMS cases reported per year in the United States (US).^1,2 OS occurs most commonly in young patients between the ages of 10 and 24 years, with a higher incidence seen in males, and in those who identify as Black or Hispanic.¹ LMS occurs most commonly in older patients of both sexes, with peak incidence after 70 years of age; however, LMS of the uterus is most commonly seen in perimenopausal women between the ages of 50 and 60 years.² Male and female patients have similar incidence but different primary sites,² and Black and Hispanic patients may be more likely to present with metastatic LMS.³ Advancements in biologic and molecular understanding, as well as development of new, effective therapies for OS and LMS, have been limited over the past decades.^1,2,4

Though 20% of new cancers diagnosed in the US are considered rare (ie, < 6 cases per 100 000 per year), research of very rare cancers, such as OS and LMS, face several challenges. These challenges include: (a) delayed or incorrect diagnosis, (b) relatively small research cohorts due to rarity which can limit discovery, (c) wide geographic distribution limiting the formation of longitudinal research cohorts, (d) certain rare cancers disproportionately affect adolescents and young adults (ie, OS) who are historically underrepresented in research,^5-8 and (e) centralization of care at tertiary urban academic sites that may introduce bias or yield findings that are not generalizable.^4,9

Engaging patients directly, wherever they live, in patient-partnered cancer research may improve our understanding and treatments of rare and understudied diseases, such as OS and LMS. Supporting participant engagement and partnership specifically in rare cancer genomics research may mitigate several of the challenges that researchers face. In addition, mitigating bias and engaging diverse populations is vital to ensure broad representation reflective of the impacted populations; however, optimal strategies to recruit and retain understudied and underrepresented communities has not been well-elucidated.^10-13 Prior literature has noted that though minoritized populations are interested in participating in genetic testing and research, their participation is limited by mistrust in research, experiences with discrimination, competing demands for time and attention, and privacy concerns.^14,15

The US National Cancer Institute (NCI) established the Participant Engagement and Cancer Genome Sequencing (PE-CGS) Network, comprised of five research centers and one coordinating center, which aims to promote effective participant engagement in cancer genomics research and to accelerate discoveries in rare, understudied cancers and in cancers that arise in diverse, underrepresented populations.^11,16 Of relevance to the PE-CGS initiative is the potential for direct benefit, such as return of profiling results, as a potential motivator for participation.

As part of the PE-CGS Network,^17,18 Count Me In (CMI)¹⁹ launched the Osteosarcoma (OS) and Leiomyosarcoma (LMS) Projects,¹⁸ in partnership with the Broad Institute of MIT and Harvard and Dana-Farber Cancer Institute, whereby any patient with OS and LMS in the US and Canada can share their health information, tumor samples, and aspects of their care and survivorship experience with cancer researchers via a web-based platform. Participants were eligible to enroll in the OS and LMS Projects if they lived in the US or Canada, were able to access the CMI website, and able to review and provide electronic consent. Participants were asked to indicate the treatment location and sign a release of their health information and biospecimens to be shared with CMI and complete a short survey on their experience with OS or with LMS.

The Engagement Optimization Unit (EOU) was formed to investigate ways to optimize recruitment, retention, and return of results and to study optimal approaches for participant engagement in cancer research, in particular with patients from underrepresented populations and adolescents.¹⁸

In this study, we report patterns and themes that support participant engagement in cancer genomics research that were identified through qualitative analysis of interviews with research partners, including patients, caregivers, and advocates.

Methods

Study Design

We conducted in-depth, semi-structured interviews with research partners, including (a) patients (age 12 and older) enrolled in the OS or LMS Projects, (b) caregivers of enrolled patients, and (c) advocates who were involved in advisory groups to the OS and LMS Projects. The study protocol was approved by the Dana-Farber/Harvard Cancer Center institutional review board, Boston, MA (Protocol # 21-721; Approval Date: June 2, 2022). The reporting of this study conforms to the Consolidated Criteria for Reporting Qualitative Research (COREQ) guidelines.²⁰

Recruitment, Enrollment, Sample Size

Research partners, including patients, caregivers, and advocates, were purposefully sampled to ensure inclusion of minoritized race or ethnicity, those with less than college education, and adolescents.

Given the motivational, logistic, and structural issues we aimed to identify were relatively independent of sample size, we anticipated roughly 5-15 interview per group (patients, caregivers, advocates), or goal of 25-30 total interviews across groups, would allow thematic saturation to be reached. Thematic saturation was assessed by the model of data saturation. Data sampling and additional interviews were conducted until new information obtained from each interview was limited, allowing us to reach thematic saturation, as detailed below in our description of the qualitative analysis.²¹

Patients were eligible for inclusion as research partners if they were age >18 years, or age 12-17 years with parental consent, had a diagnosis of either OS (for the OS Project) or LMS (for the LMS Project), enrolled in the OS or LMS Project, and were English- or Spanish-speaking and -reading. Caregivers, including both parent/guardians or other caregivers, were eligible if they participated in the care of a patient enrolled in the OS or LMS Projects and English- or Spanish-speaking and -reading.

Advocates were eligible if they were involved in the leadership or operation of, or were active volunteers for, an associated advocacy group of the OS or LMS Project and were English- or Spanish-speaking and -reading. Advocacy groups associated with the OS Project included organizations such as MIB Agents (Bernard, VT, USA) and QuadW Foundation (Dallas, TX, USA). Advocacy groups associated with the LMS Project included organizations such the Leiomyosarcoma Support and Direct Research Fund (Tulsa, OK, USA) and the National Leiomyosarcoma Foundation (Denver, CO, USA). Advocates may have been patients with, or caregivers of patients with, OS or LMS and/or enrolled in the OS or LMS Project; however, this was not required for eligibility.

Enrolled patients and caregivers were contacted via email using their provided contact information, given information about the study procedures, and offered the opportunity to participate in an interview by phone or by Zoom. Patients and caregivers who accepted were contacted by email or phone to schedule an interview. Advocates were recruited with help of advocacy partner organizations, who sent email interview requests to members via their distribution list. All research partners were offered a $50 gift card.

Data Collection

Thirty-minute individual interviews were conducted via phone or Zoom and audiotaped and transcribed by research staff familiar with this project. Transcripts were not returned to participants. Interviewers were female and male, respectively, held college degrees, and employed as research assistants. They were trained using model interviews and role playing, to use probing questions and neutral responses. Research partners were asked to recount their personal experience engaging with the OS or LMS Project, including how they first learned about the OS or LMS Projects, and their experience and impressions interacting with the website and registering for the project.

Research partners were asked to identify barriers and facilitators to participation and review the website content and registration logistics in detail. Probing questions focused on roadblocks to participation, concerns raised by any presented content, and general motivations to participate in either the OS or LMS Projects. Research partners were also asked to offer their own suggestions for improvement and outreach, as well as their thoughts on specific interventions.

Interviews were planned to be one-on-one. Adolescents were given an option to be interviewed alone or with a parent/caregiver present during the interview. In both cases, adolescents were asked additional questions, including how they learned about the OS or LMS Projects, whether their participation was driven more by them or by their parent/guardian, how information about either project was shared between their parents and them, and how adolescents can be better engaged.

Data Analysis

Interview transcripts were reviewed by the research team as they were conducted to evaluate for new or additional information from each interview. Thematic saturation was reached based on a model of data saturation prior to the start of formal coding,²¹ and recruitment and data collection were stopped at this point. A deductive codebook was developed based on the general interview guide and review of the interview note forms completed after each interview.

Coding and analysis were conducted by three members of the research team using NVivo (Lumivero; Denver, CO, USA). Using the generated codebook, each interview transcript was independently coded by two members of the research team. After independent coding was completed, researchers met to discuss discrepancies and ensure consistency in the application of the codebook to each transcript.

Thematic analysis was used to identify themes and subthemes that support participant engagement.^22,23 Direct quotes from the interview transcripts were identified to represent each theme. Participants were not provided direct feedback on the findings, but their comments and feedback were used by the operational teams of the OS and LMS Project for process improvement.

Results

Research Partner Characteristics

Nineteen patients, fourteen caregivers, and fourteen advocates were approached by email. Ten patients, ten caregivers, and six advocates consented, enrolled and completed interviews. Three interviews were conducted concurrently with an adolescent patient and adult parent/guardian caregiver (per the request of the adolescent patient); one interview was conducted concurrently with an adult patient and adult caregiver. No repeat interviews were conducted.

Research partner characteristics are provided in Table 1. Patients had a median age of 51 and were between the ages of 16 and 68 years. Seventy percent were female, 50% identified as White, 10% identified as Black, 10% identified as Asian, 20% as White and Black, 10% as White and Asian, and 40% identified as Hispanic. Half had completed at least some college (50%). Adolescent patients were between the ages of 16 and 17 years.

Table 1.

Research Partner Characteristics

	Patients (all)	Patients (adolescents)	Caregivers (n = 7)	Advocates (n = 6)
	(n = 10)	(n = 3)	Caregivers (n = 7)	Advocates (n = 6)
Age: Median (range)	51 (16 – 68)	(16-17)	48 (39-53)	61 (31 - 76)
Female: N (%)	7 (70)	0 (0)	5 (72)	5 (83)
Race: N (%)
White	5 (50)	2 (67)	6 (86)	6 (100)
Black	1 (10)
Asian	1 (10)	1 (33)
White and black	2 (20)
White and Asian	1 (10)		1 (14)
Hispanic ethnicity	4 (40)	0 (0)	0 (0)	0 (0)
Education: N (%)
Some high school	3 (30)	3 (100)		0 (0)
High school degree	2 (20)			0 (0)
Some college	5 (50)		1 (14)	1 (17)
College graduate	0 (0)			2 (33)
Graduate or professional	0 (0)		6 (86)	3 (50)

Sociodemographic characteristics were available for seven of ten caregivers who were between the ages of 39 and 53 years. Due to an operational limitation in how sociodemographic data was collected and stored via survey, sociodemographic data was not available for the three parent/caregivers who were interviewed with an adolescent patient. The parents/caregiver completed the survey on behalf of their adolescent child, but were not prompted by survey to provide data about themselves. Of the seven remaining caregivers, seventy-two percent were female, 86% identified as White, and 14% identified as White and Asian. Eighty-six percent had completed graduate or professional school. Advocates were between the ages of 31 and 76 years. All were non-Hispanic White, and a majority (50%) had completed graduate or professional school.

Research partners resided in 14 US states (California, Connecticut, Florida, Illinois, Michigan, Missouri, Montana, New York, Ohio, South Carolina, Tennessee, Vermont, Virginia, Washington) and 1 Canadian Province (Alberta).

Themes

Seven themes were identified that supported participant engagement: (a) motivation, (b) respect, (c) trust, (d) inclusivity, (e) relationship (f) engagement, and (g) empowerment. Appendix A provides partial exchanges between interviewers and research partner interviewees. Representative quotes from interviewer-research partner exchanges are highlighted in text below, and also in Table 2, to show how each quote maps to a specific subtheme.

Table 2.

Themes and Subthemes Supporting Participant Engagement, With Direct Quotes

Themes and subthemes		Examples
Motivation	Altruism/service to others	“It’s not for me, but for the ones that follow me.” [patient]
	Playing a part in scientific progress	“I want to find a cure.” [patient]
	Serving as a partner in a novel initiative	“It’s telling me that I’m a partner in this.” [patient]
Respect	Appropriate time and information requested	“It was a little longer than I was expecting.” [caregiver]
	Acknowledgement of illness	“Drawing extra blood when they’re already very low is a significant physical hardship.” [advocate]
	Timeliness in communication and follow-up	“They expected to hear back right away, and I don’t think they had a realistic view of what the timeframes were with each step…” [advocate]
Trust	Privacy and confidentiality	“It’s like a Gattaca scenario [1997 movie] that you’re worried about.” [caregiver]
	Appearance of legitimacy	“Count Me in is kind of an unknown entity.” [advocate]
	Transparency	“I want to know what’s gonna happen with the research that’s generated.” [advocate]
Inclusivity	Broad representation	“There’s no mention of LGBTQ” [patient]
Inclusivity	Acknowledgement of technical barriers	“I’m blind in one eye, so I can’t see from a far distance unless I zoom in.” [patient]
Relationship	With research team operations	“You’re asking to join here, like right up front… wouldn’t want to lose someone because you’re asking too much of them without having a relationship first.” [advocate]
Relationship	With scientists	“Like once I’ve submitted my sample, having some - not feedback, but - I don’t know how to put it. But just feeling connected - some way of feeling continually connected.” [advocate]
Engagement	General engagement	“It is really motivating to hear that you could help people a lot just from it.” [patient]
Engagement	Challenges of adolescent engagement	“Those things I usually don’t involve myself with. It’s usually always on a parent or a guardian.” [patient (adolescent)]
Empowerment	Make impact	“That every experience with cancer should be counted… is really motivating to hear, that you could help people a lot just from it.” [patient]
Empowerment	Work together	“… make people feel like it’s important, we’re all working towards this common goal.” [advocate]

Research partners were motivated by several factors to participate in a novel patient-partnered research effort. These included (i) altruism or service to others (“This is a way that you can actually make it better for another – not just one person, but for many people. This is a way to profoundly use your powers for good” [Advocate]), (ii) playing a part in scientific progress (“We're all very eager to just help in any way we can to come and reach a solution, anything, groundbreaking that could help to get us to the end of this” [Patient]), and (iii) serving as a partner in a novel initiative (“That every experience with cancer should be counted… And for someone to push that research out for everyone and to get everyone involved, that is really great” [Patient]).

Research partners wanted to feel respected by the research team. This was supported through (i) timeliness in communication or follow-up (“Communication has been a weak point, and I think that could solve a lot of these problems” [Advocate]), (ii) an appropriate amount of time and information requested of participants (“But if there's ever a barrier, it's typically time” [Patient]), and (iii) an acknowledgement that illness may prevent maximal participation (“One of the things you have to remember is people that are going through this are getting brutal treatments, like cisplatin and doxorubicin, and radiation, and you’re not feeling very well… my brain was very fuzzy” [Advocate]).

Trust in the research team was identified by research partners as an important factor to support engagement. Trust was supported by (i) ensuring participants felt adequate privacy/confidentiality safeguards were in place (“I could see privacy being a concern in any research project, just because it is dealing with sensitive information, and it’s something that’s very close and sensitive to the patient” [Patient]), (ii) the research team appeared legitimate (“If I’m just like new to this, I’m like, what am I joining? I don’t understand what I’m joining. I’m joining - I’m giving you my medical records” [Advocate]), and (iii) the research process was transparent (“The other thing is making it really clear what happens down the road” [Advocate]).

Research partners wanted to feel as though the research team meant to or wanted to include people like them. Inclusivity was supported by (i) ensuring broad representation (“Like I don't really see any East Asian sort of faces, so maybe having maybe a little bit more diversity and - or younger people” [Advocate]) and (ii) by acknowledging and proactively mitigating technical barriers (“I can see if someone who’s not that comfortable using a computer – you know, maybe the older generation of maybe individuals in lower socioeconomic tiers of society, maybe they have an issue with that” [Caregiver]).

Research partners wanted to feel a relationship with not only the research team operations (“And the staying involved, I liked that one, because I do get constant emails letting me know that you're sending the kits, letting me know they've gotten the kits, what the next step is. So I'm not left in the dark as to what's happening and how it's going” [Patient]), but also the scientists using their health information for research (“I think for me, it needs to have a warm feeling of sharers and a collaborative, not disconnected, nature” [Advocate]).

Research partners also wanted to feel engaged and empowered by the research team. Engagement was supported by promoting and supporting partnership between participant and the research team (“They really meaningfully asked for feedback and information” [Patient]). Engagement was found to be challenging specifically for adolescent patients. Of the adolescent patients interviewed, all reported their parents were more engaged in the research process than they were, which may have limited their personal engagement. Empowerment was supported by having or helping participants feel they were (i) making impact (“it’s not just for me, but for the ones that follow me” [Patient]) and (ii) working together for a common good (“make people feel like it’s important, we’re all working towards this common goal” [Advocate]).

Discussion

Though participant engagement is critical to cancer research, and patients are interested in directly partnering in research efforts, several factors challenge or prevent research teams from honoring their wishes.^11-13 Some of these factors are system-level or institution-level, such as difficulty obtaining health information and tissue biospecimens within a fragmented and siloed healthcare system and lack of interoperability among electronic health records.¹² Other factors are participant-level, such as the verbosity and technical jargon of consent forms – often available only in English — and technologic barriers and the digital divide.¹²

Patient-partnered research teams may not have agency to influence system- and institution-level factors; however, they have greater agency to influence participant-level factors through actions, behaviors, and proactive communication with participants that can either promote or hinder participant engagement. In this study of research partners, including patients, caregivers, and advocates in a novel patient-partnered cancer genomics research study, we confirmed a strong desire to engage directly in research. We also identified seven themes to support participant engagement: (a) motivation, (b) respect, (c) trust, (d) inclusivity, (e) relationship (f) engagement, and (g) empowerment. To support participant engagement, research teams can optimize their actions and behaviors and leverage these themes to improve their communication and operations to support full engagement.

Our findings allow us to speculate about potential solutions and interventions. Effective and proactive communication between researchers and participants can help participants feel motivated, engaged, empowered, and develop a relationship with the research team. In addition, effective communication can mitigate specific concerns highlighted by minoritized populations that limit their participation in genetics and personalized medicine research, such as mistrust in research and concerns about privacy.^14,15

Researchers can consider, for example, updating participants about relevant research findings using language and explanations that are accessible to a wide group. These efforts have potential to showcase to participants how their health information directly supported scientific discovery and therapeutic advances for future patients, help participants feel like their cancer experiences “counted,” and forge a bidirectional relationship between scientists and participants. Similarly, research teams can respond to concerns about privacy and confidentiality by proactively sharing information about privacy safeguards. Design elements can be used to create a public-facing aesthetic that confers legitimacy and inclusivity, along with interactive tools that support transparency. Finally, providing age- and developmentally-appropriate material on the platforms that adolescents use, such as social media, can promote engagement with this population.

Effective operations can also be leveraged to help participants feel respected and included. For example, timely communication and clear procedures and protocols can help participants feel their time and efforts are being respectfully and appropriately used. This would also support engagement of minoritized populations who have previously shared that competing demands for time and attention limit their participation in cancer genomics research.^14,15 Providing flexibility in how participants can participate, for example by allowing for options that do not involve mobile or internet technologies, can help participants feel included regardless of their technical abilities or access to digital technologies. Requesting, and iteratively responding to, feedback can help participants feel engaged and respected in the process. For example, creating a research partner advisory board that includes adolescent representation may be one way to support adolescent participants in feeling they have agency and voice, even if their parents may be leading the decision-making process.

Engaging a diverse population could also be helped by providing direct benefit to participants. Minoritized populations have previously noted that potential direct benefits are important considerations in deciding whether to participate in a given cancer genomics study.^14,15 Researchers do bear certain responsibilities to ensure the health and well-being of participants, such as prompt return of actionable results to a participant’s care, as well as the needs of the diverse communities from which participants hail.¹⁴ In addition, the benefits of cancer genomics research may be inequitably distributed both in terms of future drug discovery and potential access to novel therapeutics.¹⁴

Though direct benefit in this study was limited to planned return of research results to participants, we did find that research partners were motivated by the potential for future cancer treatments and to be engaged in research that improves not only their care, but also the care of present and future patients with their rare cancers. Inequitable distribution of research findings and access to future cancer treatments downstream of participation could be deterrents. Although we did not find this in our analysis, this could be because we interviewed research partners who were already enrolled or engaged with the OS and LMS Projects.

Our study has several limitations. Patients were invited to participate only if they had enrolled in the OS or LMS Projects, two web-based digital platforms focused on cancer genomics research, which could lead to demographic or socioeconomic bias, as well as selection bias of research partners who feel most invested in patient-partnered research. This was mitigated through purposeful sampling to ensure inclusion of minoritized race or ethnicity, those with less than college education, and adolescents. Future studies including the perspectives of individuals with minimal or no participation in a web-based cancer genomics research study could offer insights into additional and broader barriers to engagement in cancer genomics research. All adolescents chose to be interviewed in presence of their parents, even though they were offered opportunity to participate without a parent present; this may have impacted their responses. In addition, though the adolescent age range spanned 12 to 17 years, participants were between 16 and 17 years, which may limit generalizability of findings to younger adolescents. Though the sample included patients with a range of sociodemographic characteristics, the majority of caregivers and advocates were female, White, and had obtained graduate or professional education. This could be mitigated by broadening outreach to advocates beyond those who volunteer for non-profit organizations and by including other advocates, such as those identified through community outreach or social media influencers who advocate for specific patient populations.

Conclusion

Engaging patients directly in cancer genomics research is critical to improve our understanding and treatments of rare and understudied diseases, such as OS and LMS. By investing in proactive communication with patients and developing responsive operational processes, research teams can support engagement by ensuring research partners, who are motivated to participate, feel included, respected, and empowered in shared pursuit to make scientific discovery and therapeutic advances in rare cancers.

Supplemental Material

Supplemental Material - Supporting Participant Engagement in Cancer Genomics Research in Rare Cancers: A Qualitative Study of Patients, Caregivers, and Advocates

Supplemental Material for Supporting Participant Engagement in Cancer Genomics Research in Rare Cancers: A Qualitative Study of Patients, Caregivers, and Advocates by Vinayak Venkataraman, Lily Weber, Lauren Fisher, Andrew Khalaj, Eirian Siegal-Botti, Diane Diehl, Katherine A. Janeway, Suzanne George, Jennifer W. Mack in Cancer Control.

Footnotes

ORCID iDs

Vinayak Venkataraman

Diane Diehl

Ethical considerations

The study protocol and informed consent forms were approved by the Dana-Farber/Harvard Cancer Center IRB, Boston, MA (Protocol # 21-721; Approval Date: June 2, 2022).

Consent to participate

All participants provided written informed consent to participate in this research study. All qualitative data generated from semi-structured interviews was de-identified and stored in a secure, HIPAA-complaint database with access restricted only to study team members listed on IRB protocol.

Funding

The author(s) disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was conducted as part of the Count Me In PE-CGS Center, which is funded as one of five research centers by the Participant Engagement and Cancer Genome Sequencing (PE-CGS) network (Project # 1U2CCA252974-01: “Count Me In: Partnering with patients to define the clinical and genomic landscape of rare aggressive sarcomas in children and adults”).

Declaration of conflicting interests

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Data Availability Statement

Subject to appropriate ethical and legal considerations, we are open to sharing our de-identified data as relevant and necessary.*

Prior presentation

Preliminary analysis of this work has been presented as a rapid-fire presentation at the PE-CGS Network Annual Meeting in St. Louis, MO on October 9, 2024, and as a poster presentation at the Connective Tissue Oncology Society (CTOS) Annual 2024 Meeting in San Diego, CA on November 12-16, 2024. This work has not been published previously and is not under consideration for publication elsewhere.

Supplemental Material

Supplemental Material for this article is available online.

Appendix

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Supplementary Material

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Supporting Participant Engagement in Cancer Genomics Research in Rare Cancers: A Qualitative Study of Patients,Caregivers,and Advocates

Abstract

Introduction

Methods

Results

Conclusions

Plain Language Summary

Keywords

Introduction

Methods

Study Design

Recruitment, Enrollment, Sample Size

Data Collection

Data Analysis

Results

Research Partner Characteristics

Themes

Discussion

Conclusion

Supplemental Material

Supplemental Material - Supporting Participant Engagement in Cancer Genomics Research in Rare Cancers: A Qualitative Study of Patients, Caregivers, and Advocates

Footnotes

ORCID iDs

Ethical considerations

Consent to participate

Funding

Declaration of conflicting interests

Data Availability Statement

Prior presentation

Supplemental Material

Appendix

References

Supplementary Material