Engaging young African American women breast cancer survivors: A novel storytelling approach to identify patient‐centred research priorities

Abstract Background Patient‐centredness is considered an essential aspiration of a high‐quality health‐care system, and patient engagement is a critical precursor to patient‐centred care. Objectives To engage patients, health‐care providers and stakeholders in identifying recommendations to address research and practice gaps that impact young African American breast cancer survivors. Methods This paper reported an approach for research priority setting. This approach applies an engagement process (January‐September 2018) of using patient and stakeholder groups, patient storytelling workshops and a culminating storytelling conference in Wisconsin to generate relevant research topics and recommendations. Topics were prioritized using an iterative engagement process. Research priorities and recommendation were ranked over the conference by counting participants’ anonymous votes. Results One hundred attendees (43 patients/family members, 20 providers/researchers and 37 community members) participated in the conference. Five topics were identified as priorities. The results showed that three priority areas received the most votes, specifically community outreach and education, providing affordable health care and engaging in complementary care practice. Stakeholders also agreed it is critical to ‘include youth in the conversation’ when planning for cancer support and educational programmes for caregivers, friends and family members. Conclusion Storytelling as a patient engagement approach can build trust in the patient‐research partnership, ensure that patients are meaningfully engaged throughout the process and capture the diversity of patient experiences and perspectives.


| INTRODUC TI ON
Breast cancer is the most common cancer diagnosed in women and the second leading cause of cancer-related death in US women.
Most breast cancers are diagnosed in post-menopausal women, but approximately 11% of all new breast cancer cases in the United States are found in women younger than 45 years of age. 1 Young women with breast cancer may face unique challenges related to relationships, parenting, finances and employment compared to their older counterparts. Evidence suggests that young African American female breast cancer survivors (YAABCS) face even greater challenges. The breast cancer mortality rates are three times higher in young African American women than in young Caucasian women. 1 Additionally, YAABC survivors are more often diagnosed with biologically more aggressive disease (ie triple negative) and/or metastatic disease leading to poorer prognosis, aggressive treatments, long-term treatment-related side-effects and unique psychosocial concerns. In 2018, the New American College of Radiology (ACR) and Society of Breast Imaging (SBI) published breast cancer screening guidelines that were the first to recognize that African American women are at high risk of developing breast cancer and need further consideration for earlier (ie screening to begin prior to age 40) and/or more intensive screening. 2 In addition, it is well documented that Black women are more likely to experience delays in follow-up to abnormal mammography and treatment initiation, [3][4][5][6] which may be particularly salient for YAABCS. Despite these disparities, few efforts have sought to bring YAABCS, health-care providers and researchers together to inform research priorities that will lead to improved breast cancer outcomes and care.
Patient engagement is an established strategy that can be used to inform research to address health disparities and improve the delivery of effective and responsive health-care services. [7][8][9] Regardless, there is limited consensus on how best to engage patients. 7 The overarching goal of the current study was to bring together YAABCS, health-care providers and stakeholders to identify recommendations to address research and practice gaps that impact YAABCS. 10 This study contributes to the literature on patient engagement in several ways. First, it contributes to the literature on providing a detailed process to address research priority setting for YAABCS, as the literature suggests that priority setting between researchers and stakeholders may improve research relevance and value. 11,12 Second, we use an innovative patient-centred storytelling approach to effectively engage YAABC survivors. Storytelling is deeply rooted in African American culture. 13 Storytelling in the patient's own voice has the power to directly and more effectively confront a patient's health concerns, outcomes of interests, as well as barriers to receiving care across the cancer control continuum. 10,14 This approach was used in Henry Ford Health System (Detroit, Michigan, USA), which successfully led to the development of a set of patient-centred comparative effectiveness research questions. 15 Third, we apply key patient/stakeholder engagement principles and best practices from the Patient-Centered Outcomes Research Institute (PCORI). 16 Herein, we present the collaborative learning process and findings from the African American young breast cancer survivor storytelling project that culminated in a storytelling conference. The aims of this project were to: (a) learn the health concerns, outcomes of interests, as well as barriers to receiving care across the cancer control continuum from YAABC survivors through storytelling; (b) generate a list of research questions as well as practice and policy recommendations to address cancer disparities among young African American women; and (c) facilitate new relationships among patient, researchers, clinicians and policymakers.

| Study design
This study reported an approach for research priority setting. This approach we outlined in the paper applies an engagement process of using patient and stakeholder groups, patient storytelling training workshops and a culminating storytelling conference to generate relevant research topics and questions to improve breast cancer care in YAABCS.

| Recruitment
We established two groups: (a) a patient advisory group of 10 female YAABC survivors; and (b) a stakeholder group comprised of six members, including health-care providers, academic researchers and representatives from patient advocate groups currently engaged in topics related to breast cancer and/or breast cancer disparities.
Patients (YAABC) were eligible if they were African American women the official views of the Patient-Centered Outcomes Research Institute (PCORI).

Conclusion:
Storytelling as a patient engagement approach can build trust in the patient-research partnership, ensure that patients are meaningfully engaged throughout the process and capture the diversity of patient experiences and perspectives.

K E Y W O R D S
African American, patient engagement, patient-centred approach, storytelling, young breast cancer survivors living in Milwaukee County with a history of breast cancer diagnosed between the ages of 20 and 45 years old. We used the age of 45 as the cut-off to define young breast cancer patients based on the CDC's definition. 1  Three approaches were used to recruit participants and conference attendees: (a) physicians and nurses recruited patients during their clinical visits (patients only); (b) existing community-based partnerships were leveraged to recruit through breast cancer support groups, local African American beauty salons, churches, neighbourhood health centres and social service agencies; and (c) advertisements and programming on V100.7, a popular radio station in the African American community, provided information on the opportunity to join the patient or stakeholder group and/or attend the storytelling conference. The study was exempt from the Institutional Review Board as it only involves the engagement activities related to a conference.

| Stakeholder group meetings
Between March and May 2018, our research team convened two key discussion groups: one with the patient advisory group and one with the other stakeholders to generate and refine topics to be discussed in the upcoming storytelling conference. For each group, we asked 'what are the most relevant and important questions/issues across the cancer control continuum (from prevention to survivorship) that you believe would improve breast cancer outcomes among young African American women'. We also solicited patient-related concerns and challenges (eg screening, diagnosis, treatment and survivorship) among the patient advisory group. The topics from these groups were summarized by themes, which were then voted on at the beginning of the storytelling conference for further discussion among conference attendees. Our reimbursement practice adhered to PCORI's best practice on 'compensation and reimbursement of out-of-pocket expenses'. 16

| Storytelling workshops
Prior to the storytelling conference (May-August 2018), we conducted a series of six storytelling workshops. The purposes of the workshop were to identify the top five concerns and challenges of African American breast cancer survivors and to help survivors identify, structure, practice and share their personal stories effectively. To make the storytelling process more concise and captivating for the audience, we partnered with a local non-profit storytelling organization, Ex Fabula, and hired an African American storytelling coach to facilitate the workshops. The coach managed six 120-minute conversational style workshops for the 10 participants, during which participants were taught the basics of storytelling and ways to be successful in the art of storytelling. Workshop topics included identifying their challenges and concerns, creating story ideas (to illustrate these challenges and concerns) and story structure, determining conference agenda (ie which stories, what order, presentation time) and others. Upon completion of the workshops, participants were invited to record their stories. These stories, which highlight the need to develop evidence-based interventions and policies, were posted on YouTube with participant consent.

| Conference overview
The storytelling conference titled, 'Sisters We Thrive, Stories We Tell', was scheduled on a Saturday from 8:30 am to 2 pm in Milwaukee, Wisconsin. YAABC survivors, their family members or caregivers, health researchers, clinicians, representatives from patient advocate groups and other community stakeholders were invited. The conference included keynote speakers, storytelling and discussion groups.
To inform discussion group topics, attendees received a short questionnaire upon arrival that requested information on their identified gender and race/ethnicity. The conference attendees were also asked to rank by order of importance of the eight research topics/ themes generated by the stakeholder meetings and storytelling workshops (Table 2). Then, conference staff summarized the ranking. Of the eight topics related to improved care and outcomes for YAABCS, five were ranked as most important ( Table 2). The conference kicked off with a general session of keynote presentations by three local health disparity researchers, followed by a two-hour StorySlam (ie storytelling event) 19 coordinated by the storytelling coach. The ten participants who participated in the storytelling workshops shared their stories with the conference attendees. A variety of formats was used including: five longer stories, short anecdotes and UltraShorts. 20,21 Other conference attendees had the chance to participate actively at the event, if they desired, by writing a few sentences on a piece of paper based on reflections and attendee stories and having them read on stage as UltraShorts.
Following the storytelling, attendees were encouraged to join one of five discussion groups targeting a particular topic that was voted as high priority at the beginning of the conference. Each group had a facilitator who followed a five-step protocol (Table 1)

| Patient advisory and stakeholder group: generating research topics
The stakeholder meetings (with the patient advisory group and the stakeholder group) and the patient-centred storytelling workshops generated a list of eight research topics most relevant to YAABC survivors. Those topics (shown in Table 2) were used as the starting point for further discussions in the storytelling conference.

| Storytelling conference: ranking topics
Of the eight topics related to improved care and outcomes for YAABCS that were generated by the stakeholder meetings and storytelling workshops, conference attendees selected five. The most highly ranked topics were ( Addressing access to care and insurance coverage, such as eligibility for early detection (based on age), access to quality mammography, referrals to genetic counseling 8

(#1)
Providing support and educational programs for caregivers, friends, and family members of breast cancer survivors 7

(#2)
Managing breast cancer treatment side effects (tiredness, headaches, pain and numbness, lymphedema, bone loss and osteoporosis, heart problems, menopause, sexual difficulties, infertility, chemo brain), and addressing their impact on quality of life and minimizing the impact of financial hardship experienced by cancer survivors 6

(#3)
Enhancing patient understanding of treatment options (standard of care, clinical trials) and right to seek second opinions 5

(#4)
Incorporating spirituality and positive thinking during and after treatment 4

(#5)
Learn how to better cope with changes in personal life during and after treatment (relationships, the possibility of early menopause caused by chemotherapy, fertility, sexuality, psychological distress, disruption of employment, others)  Table 3). Table 3; Figure 1)

| Final voting on recommendations and solutions: Comparing and contrasting priorities among different stakeholders (see
The colour dot voting provided a unique opportunity for different stakeholders (patients/family members, researchers/health professionals and community members) to decide what matters most when addressing health disparities among YAABCS. Figure 1 shows the numbers of vote for each statement by three different types of stakeholders.

| D ISCUSS I ON
Patient-centredness is considered an essential aspiration of a high-quality health-care system, 23 and patient engagement is a critical precursor to patient-centred care. 7,24 As concluded by the 2014 Institute of Medicine: 'Prepared, engaged patients are a fundamental precursor to high-quality care, lower costs and better health'. 25 In this study, we provide an example of how to actively engage patients and other stakeholders using established best practices, along with a novel storytelling approach. 16 The engagement approaches including storytelling contribute to the literature on using innovative methods to address the knowledge-to-action gap 26 between the 'what' and 'how' of patient engagement. 7 Patient engagement can happen at several levels from inform, in which little or no active participation occurs (eg receiving information via a website or booklet), to empower, in which the goal is to provide patients the necessary tools to feel comfortable with decision-making power. 27 Several measures were put in place to ensure genuine and meaningful engagement of patient stakeholders.
First, patients were closely involved throughout the research topic generation and prioritization process. Trust was built early between stakeholders and research team through regular dialogue.
A fun and interactive storytelling approach empowered patients to use their own voice to directly and more effectively confront their health concerns, outcomes of interests, as well as barriers to receiving care across the cancer control continuum. 10,14 Second, patient stakeholders had the decision-making power. A shared and collaborative decision-making process was used throughout the entire project period to identify research priorities. 16 We ap-  insurance, fear of mammogram and treatment delay due to fear of death or lack of insurance all contribute to disparities at the patient level. At the provider level, our patients' stories showed that some doctors may ignore or inadequately address minority women's serious side-effects of treatment. Our finding also highlighted the importance of addressing patient-provider communication factors 29,30 such as training providers as good listeners and addressing patients' spirituality needs when necessary. 31 Health system factors such as higher copayment requirements, lack of a routine source of care, distance to care, fragmentation of care and uneven distribution of screening and treatment resources also exacerbate disparities. 28 This approach, although similar to other studies, 9,38 may not be ideal for increasing interactions between researchers, clinicians, and patients.
Findings have the potential to inform research, practice and policy in addressing health disparities among African American young breast cancer survivors. We anticipated that the next steps are twofold. First, the statewide policymakers, practitioners and decision-makers within local health-care systems need to be informed about our findings. The research team has already started the dissemination process. For example, our findings as well as the patient storytelling film were featured and discussed at the statewide cancer summit and health-care delivery planning meetings. Second, final priorities will be disseminated with researchers so they can utilize this information to guide the design of future research projects. This conference established partnerships between patients, providers and researchers. Together, we plan to investigate many of the priorities outlined here with large comparative effectiveness research studies.
Patient engagement has the potential to enrich our understanding of patient priorities for research. Given the current focus on developing patient-centred research questions, we suggest future studies to vigorously define and evaluate various patient engagement approaches and determine what approach works best, under which circumstances. Successful approaches will build trust in the patient-research partnership, ensure that patients are meaningfully engaged throughout the process and capture the diversity of patient experiences and perspectives. The detailed patient engagement approach we used in this study provides a blueprint for groups interested in pursuing a collaborative approach in which multi-stakeholder groups work together to identify research priorities.

ACK N OWLED G EM ENTS
The authors would like to acknowledge the Jennifer MP Woo, MPH, Mary Elise Papke, PhD, and Ms Lee Goodwin, Megan McGee and Kennita Hickman from Ex Fabula for their assistance. We also want to thank all survivors and participants who participated in the storytelling workshops and conference.

CO N FLI C T O F I NTE R E S T
All authors declare that he/she has no conflict of interest of this study.

DATA AVA I L A B I L I T Y S TAT E M E N T
The data used to support the findings of this study are available from the corresponding author upon request.