From Patient to Participant: Enhancing the Validity and ... - Springer Link

J Canc Educ (2013) 28:237–246 DOI 10.1007/s13187-013-0464-2

From Patient to Participant: Enhancing the Validity and Ethics of Cancer Research through Participatory Research Connie G. Chiu & Terry L. Mitchell & Margaret I. Fitch

Published online: 20 April 2013 # Springer Science+Business Media New York 2013

Abstract Participatory health research involves a wide spectrum of participation from the population of study. We describe the participatory research processes of a large mixed method study on the psychosocial impact of dragon boating in individuals with breast cancer. In particular, we discuss the involvement of a Community Advisory Group (consisting of five breast cancer patients/survivors) in the development of the research study, data collection and analysis, and dissemination of the study results. We also outline the elements of a research workshop, in which 13 breast cancer patients/survivors were involved in the development of a provincial survey for the study. The purpose of this article is to share our experience of engaging cancer patients/survivors in a participatory research study. We discuss the value-based elements of participatory research (power sharing, voice and respect, reciprocity, and mutual benefit), and provide a case-based example of how these participatory elements were employed to potentially increase the validity of the survey instrument, to enhance the ethics of working with a cancer population, and ultimately contributed to a high survey response rate.

C. G. Chiu (*) Surgical Oncology, John Wayne Cancer Institute, 2200 Santa Monica Boulevard, Santa Monica, CA 90404, USA e-mail: [email protected] T. L. Mitchell Department of Psychology, Wilfrid Laurier University, 75 University Avenue West( ON Waterloo N2L 3C5, Canada e-mail: [email protected] M. I. Fitch Odette Cancer Centre, Sunnybrook Health Sciences Centre, 2075 Bayview Ave., Room T2 234( ON Toronto M4N 3M5, Canada e-mail: [email protected]

Keywords Community based participatory research . Participatory research . Breast cancer . Psychometric instruments . Patient involvement

Introduction Over the past two decades, there has been an increasing presence of community-based research and participatory methods in health research [1–3]. Involving patients at various stages of the research process has been shown to improve the ethical nature of research and the appropriateness of methods [4–6]. The involvement of cancer patients in conducting cancer research has been discussed as a means of developing the cancer care system to become more responsive to the needs of people living with cancer [7]. Involving patients in cancer research has been advanced by psychosocial researchers such as Wright, Corner, Hopkinson, and Foster as an imperative in cancer studies [8]. Despite the benefits of involving cancer patients as consultants in cancer research, a review of the literature regarding the involvement of people affected by cancer in research revealed only 57 relevant articles published between 1994 and 2010 from the USA, UK, Canada, and Australia combined. Of these studies, only nine employed a participatory research (PR) design. The purpose of this article is to contribute to the literature on patient participation by sharing the details of a breast cancer study in which cancer patients/survivors were engaged in all stages of the study from question formation to conference presentations and study publication. We use the combined term patients/survivors to indicate the compromised health and medically engaged nature of this population while respecting those who prefer the term survivor to indicate that they are reclaiming power, agency, and hope in the aftermath

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of the disempowering processes of their cancer diagnoses and treatment. In this paper, we report on the extensive PR processes we utilized to conduct a study on the psychosocial impact of participation in breast cancer survivor dragon boating. We share the specific ways we engaged people living with cancer in our research as members of an ongoing Community Advisory Group (CAG) throughout the study and in particular, as research consultants in the development of a provincial survey. In this article, we present a background on participatory processes in health research, a description of the survivor dragon boat study, then an outline of the specific participatory processes of this study. We conclude with a discussion of the benefits and risks of increased participation of cancer patients/survivors in designing and conducting psychosocial cancer research and offer accommodations to strengthen both the validity and the ethics of psychosocial cancer research through participatory processes.

Background Participatory Research PR is an approach to research that seeks to create an equitable and non-oppressive experience for research participants and stakeholders with a focus on the co-creation of knowledge [9, 10]. In PR, a collaborative relationship is formed between researchers and those affected by the phenomenon being studied with stakeholders being actively involved in advisory committees and/or as research assistants. PR researchers serve as facilitators and catalysts to enable members of the research population to contribute their lived knowledge and experience to inform and enhance the research process [3]. Particular consideration is placed on the research relationship between researcher and research participants with attention to issues of power and voice [3, 11]. PR involves bidirectional learning and mutual benefit for both researchers and participants [12]. There is a shift from the role of research subject or passive data source to advisor or coresearchers in one or more phases of the research, including the development of funding applications, research designs, data collection, interpretation, and/or dissemination [13]. Rationale for Patient/Survivor Participation While there has been very little published on the research engagement of cancer patients, a few studies have reported on the benefits of including people living with cancer in the selection of domains and the development of instruments to improve the content validity of quality of life (QOL) measures [14, 15]. Kindree et al. utilized focus groups to assess the comprehensiveness, ease of understanding, feasibility of items,

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and survey implementation issues of a newly developed cancer screening instrument [16]. They assert that survivor participation in the review of instruments is more effective than the more traditional research process of piloting instruments. In conducting psychosocial breast cancer research, there is an increasing need to understand long-term QOL issues. Psychometric scales have been a valuable tool; however, the selection of domains of interest in QOL measures and the development of such surveys have been largely expert driven to the exclusion of the experiential knowledge of patients/survivors themselves [15]. As a result, many measures focus on questions of post-treatment basic function that may be inappropriate for longer-term survivors. Further, existing QOL measures have been criticized for failing to capture the experiences of cancer survivors, and for focusing on deficits rather than the personal knowledge and growth gained from their illness experience. Lacking adequate participation of patient/survivors in instrument development and review has resulted in QOL instruments that have often excluded psychological and social dimensions. It has also been noted that very few measures address existential issues, such as spirituality and life meaning; issues that are critical to many cancer survivors [14, 17]. In our work, we attempted to address these instrument deficits by engaging cancer survivors in the review and selection of previously validated psychometric measures to more accurately reflect and assess the psychosocial impact of cancer diagnosis, treatment, and participation. Survivor Dragon Boat Study The Ontario Breast Cancer Survivor Dragon Boat Study was a participatory, mixed method, community-based research project that examined the experiences and psychosocial impact of dragon boating in the lives of women who had been treated for breast cancer. Survivor Dragon Boating was introduced into Canada through the research on lymphedema and upper body exercise [18]. Dragon boating is based on traditional Chinese long-boating, and involves 22 individuals working synchronously to advance a 40–60 ft boat in races of 2–4 min duration. Survivor dragon boat teams consist of members that have been diagnosed and treated for breast cancer. While research has been conducted with survivor dragon boat teams on the physiological effects of strenuous upper arm exercise [18–20] and the promotion of exercise [21], our work focused on the study of the psychosocial impact of survivor dragon boating [22–24]. The research team was comprised of the principal investigators and a CAG that involved five breast cancer survivors who were leaders in their survivor dragon boat teams. The CAG was involved at all stages of the study, including research design, funding proposal, participant recruitment, qualitative interview guide development, review

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of findings and preliminary analyses, workshop design, survey development, and the presentation and publication of findings, including national conference presentations, journal articles, and a book chapter [22–24]. Of the three refereed publications, the book chapter consists of two survivor authors and an article with a survivor co-author. The extensive participatory processes of the Survivor Dragon Boat Study will be described below in consecutive phases of this mixed method study that began with extensive in-depth qualitative interviews with 56 patient/survivor dragon boaters and culminated in a large provincial survey of 405 respondents. The participants ranged in age from mid-20 to mid-80 years with a mean age of 55 years. The women had received a variety of treatments for breast cancer including radical mastectomies, chemotherapy, radiation, and hormone replacement therapy. The respondents reported a significant burden of disease with 17 % of individuals diagnosed at stages 3 or 4 [22].

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from this first set of data were reviewed together by one of the survivor dragon boaters involved in the pilot interviews and the principal investigator. This participant then collaborated on and co-authored the first publication from the study along with the principal investigator [24]. The findings of the pilot interviews were used to inform the next stages of the research and grant development. Community Advisory Group All six women who were interviewed in the pilot study were invited to serve as members of the CAG for the Survivor Dragon Boat Study. Five of the six women from the pilot interviews formed the CAG for the duration of the study, and continued to guide all phases of the research planning, implementation, analysis, and dissemination processes on a quarterly basis for the next 3 years. The CAG provided consultation and guidance at all phases of the study including the review of interview guides, initial findings, and all community processes for participant communication and recruitment.

Qualitative Phase In-depth Interviews Pilot Interviews The Survivor Dragon Boat Study began with six in-depth qualitative pilot interviews with breast cancer patient/survivors. Qualitative research generally employs small purposeful samples that are selected to provide indepth data on a phenomenon of interest. In purposeful sampling (Patton 1990), the sample should be judged on the basis of the purpose and rationale for the study and the sampling strategy used to achieve the studies purpose. Three members from each of two different teams (located in Toronto and Thunder Bay) were chosen to provide geographical variance for the study. We sought to incorporate the experiences of women from a large urban center at the south of the province (Toronto,) and a small rural center in the northern region of the province (Thunder Bay). The women were recruited though criterion based sampling in which individuals with breast cancer who were team captains of these survivor dragon boat teams were contacted for interviews. These individuals each submitted the name of two other team members who were interested in being interviewed, one member below the age of 45 years, and one member greater than 45 years of age at diagnosis. Despite the small sample size, there was considerable variance in racial/cultural backgrounds, education, income, relationship status, as well as a geographical and age differences. Face to face open-ended interviews were conducted with a 12-question interview guide. Participants were asked questions designed to elicit descriptions of: their experience of dragon boating and the physical, emotional, mental, and social impact of dragon boating. The findings

A series of qualitative interviews were conducted to build on the results and emergent themes of the pilot interviews and to deepen our understanding of the survivor dragon boating experience. The additional interviews involved 46 survivor dragon boaters. After the pilot interviews were completed, it was clear that the issue of psychosocial impact of survivor dragon boating was an important area of investigation that would yield rich data on physical, mental, emotional, benefits, and postdiagnosis quality of life. We therefore sought to recruit women to participate in the interviews who had a range of experiences. We attempted to recruit women representing all 13 dragon boat teams in the province from diverse groups, resulting in involvement of new members (10 participants), seasoned members (23 participants), and team leaders (13 participants). The multiple phases of data collection informed one another in an iterative process in which the interview guides were adapted for each successive phase of the study. The interview transcripts were analyzed in an ongoing manner for emergent themes that represented the experience, issues, and insights of breast cancer survivors who were participating in survivor dragon boating across Ontario. The main themes arising from the qualitative analysis were grouped categorically into four distinct domains of interest (quality of life, transcendence, psychosocial well-being, and post-traumatic stress) as the basis of the content focus for a follow-up provincial survey. The CAG met to review the data and confirmed the categorization and the four selected domains of interest.

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Quantitative Phase Identification of Psychometric Instruments The CAG group was central in conceptualizing, planning, and implementing the patient/survivor research workshop to triangulate the in-depth qualitative data set (52 interviews) with a provincial survey (405 participants). The literature was reviewed for all validated psychometric instruments that had been utilized with a breast cancer population. Those instruments that matched with the identified domains of interest (quality of life, transcendence, psychosocial well being, post-traumatic stress) from the qualitative phase were systematically reviewed and evaluated by the research team based on the following criteria: (1) Meaning: the relevance of the questions to survivor dragon boating and the cancer experience; (2) Impact in the Literature: the information gained from the use of the instrument in previous studies; (3) Instrument: Characteristics: the validity and reliability of the instruments and the time and ease of completion; and (4) Conceptual fit: the manner in which the instruments would interact and complement each other. The research team decided that Ferrel’s Quality of Life Scale [25] would be presented for the domain of QOL, as the instrument had been developed through qualitative interviews with breast cancer survivors and had been revised to include questions regarding spirituality. This scale would, however, be reviewed by the patient/survivor dragon boaters for suitability for inclusion in the study survey package. Two previously validated scales were identified for the remaining three domains, transcendence, psychosocial well-being, and post-traumatic stress, for a total of six psychometric instruments. The instruments were the Purpose in Life Test [26] and the Self-Transcendence Scale [27] for the domain of transcendence, the PTSD Checklist [28], and the Impact of Events Scale [29] for the domain of Post-Traumatic Stress, and the Mental Adjustment to Cancer Scale [30], and the Profile of Mood States [31] for the domain of psychosocial well-being. The seven identified instruments were presented to the five patient/survivor members of the CAG at an Advisory Group meeting. These instruments were reviewed in depth by the CAG survivor dragon boaters and approved for presentation to other participants at a patient/survivor research workshop. The CAG group then assisted in the design of the workshop and the selection and planning of the activities, and logistics of survivor communication and participation.

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five CAG members. A member of each survivor dragon boat from 13 different communities across Ontario was invited to participate in the research workshop. These same women had previously been involved in key informant qualitative interviews in the qualitative phase of the study. The patient/survivor participants were provided with complimentary airfare, ground transportation, two nights’ accommodation and meals, as well as an honorarium in recognition of their time and expert knowledge as consultants for the research workshop. The goal of the workshop was the development of a survey package to be comprised of selected previously validated psychometric measures for use in the provincial study of the psychosocial impact of breast cancer survivor dragon boating. Attending to Process in Patient/Survivor Research Participation Particular attention was given to the process of the workshop. A facilitator was utilized to manage the agenda and to attend to meeting logistics and group process, and a counselor was utilized to provide support to individuals if needed. The facilitator was a member of the research team with expertise in breast cancer and proven facilitation skills. We provided social events to enable networking among members of the survivor dragon boat teams. For example, the evening before the workshop began, the researchers hosted a community building dinner to promote comfort, to facilitate social networking, and to increase the focus and productivity of the research meeting. Throughout the workshop, session breaks, food and social interaction activities were facilitate to engage participants and to provide support and relief while addressing the topic of cancer and cancer treatment. Presentation and Confirmation of Qualitative Findings The research workshop began the following morning with a presentation by the principal investigator of the findings from the analyses of the 52 interviews from the qualitative phase of the study and the identification of the four selected domains of interest. Workshop participants were asked to respond to the presentation of the qualitative findings and to provide confirmation and/or critique of the selected domains of interest. Following the confirmation of the domains of interest, the facilitator proceeded to introduce the seven psychometric instruments that would be reviewed.

Patient/Survivor Research Workshop Review and Completion of Psychometric Instruments A day-long patient/survivor research workshop was then held in Toronto, Ontario, Canada at a hotel conference facility. The workshop involved 13 survivor dragon boaters from across Ontario with the participation and support of the

Workshop participants completed each of the instruments individually, and then selected, based on their experience and knowledge, the most appropriate instruments for each

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domain for utilization in the provincial survey package. In addition to their personal comments and reactions, the participants were asked to assess the scales on the criteria of clarity, comfort, and relevance (Table 1). Workshop participants were advised that any or all of the instruments could be rejected if they were determined to be inappropriate to their lived experiences. The instrument review from the breast cancer patient/survivors perspective was critical in assessing the validity and ethics of the survey instruments and study process. The objectives were to reduce the degree of error in survey responses, to increase the reliability and validity of the questions, to increase survey completion rates, and to increase the ethical nature of the research by attending to research participant experience, burden, and risk. Discussion Clarity, Comfort and Relevance of each Instrument After individual review, completion, and rating of each scale, the participants discussed their reactions to opinions of the instruments in groups of four to five individuals, with members of the CAG as group facilitators. The small group discussions were followed by a facilitated discussion with all the workshop participants and the principal research investigators. Each instrument was systematically discussed by the group utilizing the criteria of clarity, comfort, and relevance. The results of the discussion are summarized in Table 2. Instrument Selection Participants were given the ability to reject or accept each instrument for utilization in the provincial survey package through a methodology called “Dotmocracy” [32]. Dotmocracy is an equal opportunity facilitation method aimed at recognizing levels of agreement in large-group decision-making. Each individual voice is represented and considered in arriving at the group’s collective preference. It was first described by Jason Diceman and has been popularized at the National Conference on Dialogue and Deliberation in San Francisco, CA, USA. Dotmocracy has Table 1 Criteria for instrument review

been recognized and utilized by mainstream health and political researchers around the world [33, 34]. In this process, each workshop participant was provided with red and green sticker dots, and the workshop participants placed a green dot on the instruments they preferred for each of the domains, a red dot on the instruments that they determined were either not appropriate or less applicable for utilization in the study. Instruments that received a majority of votes (green dots) were selected for use in the survey package. The Quality of Life Scale [25] was approved by participant consensus for inclusion in the survey for the domain quality of life. Instrument Sequencing The participants determined the sequential order of the instruments as they would appear in the survey package, considering the experience of an individual cancer patient/survivor that would receive and complete the measures alone in their home. The selected instruments and their identified sequence for the survey was: (1) Quality of Life Scale [25], (2) Mental Adjustment to Cancer Scale [30], (3) Post-Traumatic Stress Disorder Checklist [28], and (4) Self-Transcendence Scale [27], followed by a demographic questionnaire and a section for an open-ended question. Survey Distribution Participants were asked for their advice regarding the methodology for distribution of the survey package to other dragon boat team members; either delivery by the workshop participant or by mail utilizing team contact lists. This discussion was provided in recognition of the privacy and confidentiality of breast cancer survivors and to seek the advice of the key contacts from each team on the most appropriate recruitment process for their individual teams. A 141-item survey package was distributed to all survivor dragon boat team members in Ontario utilizing the Dillman Total Design survey process [35]. Following the initial survey distribution, a second survey package was sent to individuals who had not responded to the previous survey, and following this, reminder post cards were sent to remaining individuals [35]. The survey package was

Questionnaire Participants’ perspectives characteristic

Researchers’ perspectives

Clarity

Degree of error in survey responses. Reliability and validity of the questions. Ethics. Completion rates.

Comfort

Relevance

Are the questions understandable? Are the directions clear? How distressing is this instrument? How willing will women be to answer the questions? How meaningful are the questions to women with breast cancer? How relevant are the questions to dragon boaters?

Contribution to knowledge. Completion rates.

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Table 2 Summary of instrument discussion in a workshop of women breast cancer patients/survivors Instrumenta

Transcendence Purpose in Life Test (0 votes)

Self-Transcendence Scale (13 votes)

Post-traumatic stress PTSD Checklist (11 votes)

Impact of Event Scale (2 votes)

Criteria for instrument review Clarity

Comfort

Relevance

• Questions were straightforward and clear

• Some questions were too harsh and depressing • Some of the wording was awkward

• Questions were extreme- either very good or very bad feelings (negative vs positive answers) • May be geared towards an older population

• Questions were straight forward and clear • A scale of 1–7 allows participants to not truly decide their feelings (score 4)- advantage and disadvantage of this

• Comfortable, not stressful

• Instrument had particular relevance to dragon boating • Questions were down to earth

• Generally easy to understand • Some questions repetitive • Some questions had too many words—lost attention

• Generally not stressful • A few questions were uncomfortable for some participants

• Relevant • Overall drawback was focus on mental state

• Easy to read, but some questions hard to understand

• A value judgment implicit in some questions • The use of “it” in regards to breast cancer is impersonal

• More directed at recently diagnosed people, less relevant for most participants of this study • Questions were non- specific

• Instrument was meaningful

• Some questions quite stressful to complete Psychosocial well-being Mental Adjustment to Cancer Scale (11 votes)

Profile of Mood States (2 votes)

Quality of lifeb Quality of Life Scale (13 votes to accept, 0 votes to reject)

• Instrument is long—a weakness • Scale somewhat confusing to use at first; but fine once you get used to the scale

• Questions were comfortable

• Able to bring out more feelingsthis was viewed as a strength by the participants • Questions were insightful and appropriate

• Questions were short • Easy to respond

• Negative wording- too much negativity in questions

• Relevant but not comprehensive needed positive wording/questions • Would also like to talk about the positive experiences/feelings

• Answer key was easy to use. • Questions were clear and easy to understand. • Does not specify time or frame of reference for answers.

• Good balance between information gathering and comfort completing the scale; depth of questioning was sometimes stressful although not beyond what is reasonably encountered in daily life • Prefer use of cancer “survivor” rather than cancer “patient”

• Dimensions of interest were well-addressed • Instrument does not address menopausal signs and symptoms • Failed to address side effects of treatment and the use of hormonal therapy

Comments are indicated in verbatim, including comments from individual review by the participants and from group discussion among the participants a b

Number of women that voted for inclusion of the instrument in the provincial survey through a process of dotmocracy

One instrument was presented for the Quality of Life domain. The women voted to accept or reject the use of the instrument in the provincial survey

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distributed to 500 survivor dragon boaters across Ontario and a completed survey was received from 405 (81 %) individuals.

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power sharing maximized respect for and amplification of patient/survivor voice, consensus on the content of the qualitative findings, and the development and participatory implementation of the provincial survey.

Discussion Reciprocity and Mutual Benefit Participatory Research Process Participatory research is founded on the principles of power sharing, participant voice, respect, reciprocity, and mutual benefit [12, 36]. We attempted to incorporate these principles into our study and into the structure and processes of the research workshop. Participants indicated that they liked “being at the development stage” and felt a “camaraderie and a sense of connection among the participants and with researchers”. Power Sharing Power and ownership of the research process between researchers and participant members need to be clearly addressed to allow participants to discuss, challenge, or reject the opinions of principal investigators. In order to establish increased knowledge sharing between participants and researchers, workshop participants were engaged in a discussion of the qualitative interview findings. This initial conversation revealed face validity for the key themes that were to be explored in the survey and served as a vehicle to model power sharing between researchers and patient/survivor participants. Participants had the power to select or reject any of the instruments, to order the instruments, and to determine the survey recruitment strategy for their teams. Furthermore, the group had the opportunity to withdraw their participation if they believed that it was not in the best interest of their survivor dragon boat teams to participate in a provincial survey. Voice and Respect Participants were given an opportunity to comment on the qualitative findings to confirm or disconfirm their credibility and to discuss the relevance of the key domains that had been distilled from the interview data. Workshop participants were asked to review and complete the seven identified psychometric instruments, to reflect individually on them and to make notes, to discuss their ideas in small groups, and to discuss their findings with the principle investigators in the larger group forum. Participants were also given the opportunity to reject or accept each of the psychometric instruments through an active dotmocracy process. This facilitated process of engaging patient/survivors in the research process was the vehicle by which, as researchers, we had worked to facilitate and catalyze breast cancer survivors’ voice. The research workshop process, based on the principle and practice of

In working with a cancer population, we extended the principles of PR to include the elements of comfort, safety, and care. We ensured that there was adequate travel time, appropriate, and comfortable accommodation for two nights for women whose energy may be still be compromised by treatment, and adequate breaks for rest during the workshop. Furthermore, a psychotherapist who is also a cancer survivor was available for debriefing throughout the workshop to ensure that group process and individual needs were a priority in the day. The workshop agenda was balanced to allow maximal time for completion of the study tasks while allotting time for activities of comfort and care for the participants. An honorarium of $250.00 was given to each of the workshop participants as an appreciation for their time, expertise, and contributions as research consultants. The amount was rationalized and approved in the ethics review process. The honorarium was equivalent to a physician or researcher fee for participation in a focus group. We believed that participants were experts in their experience essential to the research validation and instrument development. The existence of an honorarium was not known in advance by the participants in order to mitigate undue influence for participation. Time was also provided for participants to socialize and network among other members of dragon boat teams across Ontario. Organized breaks, meals, and social activities were integrated into the workshop agenda, and the researchers incorporated elements of fun and celebration to the research process to provide relief from the intensity of the study topic. As an example, one activity involved the construction of theme related dragon figures by the participants utilizing materials provided: wires, cloths, foils, feathers, beads, and other various craft materials. The workshop participants were split into teams and the event was timed so that the groups had to “race” to finish their dragons. The patient/survivor participants displayed their creative dragons to one another, and the activity ended with much laughter, song, and photographs of women with their dragons. Mutual Benefit Patient/survivor participation in all phases of the study assisted us in achieving recruitment success in qualitative interviews and in the provincial survey involving 405 participants. The CAG improved our qualitative interview guides and guided all process of the research project. The workshop participants made an invaluable contribution to the review,

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selection, and the ordering of quantitative instruments for the survey package. The CAG and workshop participants also represented gatekeepers to the dragon boat communities. We believe that the PR methodology resulted in enhanced survey distribution and response. The typical response rate from a mail survey is approximately 17–23 %; therefore, receiving a survey response of over 80 % in our study was an overwhelming success that can be largely attributed to the in-depth participatory approach. While we did not directly investigate the reasons underlying our participant response, we postulate that the study population was very responsive to the study because of their awareness that the survey package was developed in collaboration with their survivor dragon boat team members and peers, and that the Dillman method was also an effective means of increasing survey response rates. Reciprocity Within PR and the principles of power sharing, participant voice, respect, reciprocity, and mutual benefit, the essence of collaboration is a vested interest and gain for both parties. The patient/survivors benefited through their active involvement in the research process by being engaged in a respectful manner that acknowledged and drew upon their knowledge and expertise. This level of active engagement in the research process was an opportunity to regain a sense of control of an otherwise disempowering life trajectory by contributing to cancer research that may benefit others who are diagnosed with breast cancer. The research process provided a sense of control and power of their knowledge, in their positioning as experts in the research process. Furthermore, the facilitated research workshop also provided reciprocity in the form of an honorarium and the opportunity for social networking with peers. As highlighted by quotes from workshop participants, the goal of the breast cancer patient/survivor participants was such that they themselves wanted to direct, speak, and educate the scientific community: “I am more than willing to put effort into a project if it may benefit my daughter and future generations” and “hopefully it will bring breast cancer forward—people will be able to talk about it.” Bilateral Risk As well as the inherent benefits of PR, there were also risks in this type of research collaboration for both the researchers and the patient/survivors. The sharing of power to such an extent raised the possibility of conflict and delays in the study, and may have resulted in the rejection of all of the psychometric instruments, the failure to reach consensus on the implementation of a provincial survey, and a profound loss of time and investment. Fortunately, in this case, the risks taken by the researchers promoted trust and high levels of engagement

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among key stakeholders. Rather than delaying or derailing the research project, the careful attention paid to participatory processes at all stages of the research resulted in a sound methodological process, a credible survey instrument, a high survey response rate, and knowledge outputs which were meaningful for both the study population and the researchers. One methodological impact occurred when we discovered a lower response rate for participants who received the survey package from their team member who was present at the workshop rather than through a researcher initiated mail out. The survivor dragon boaters who received the survey package from their team representatives were instructed to give back their completed packages to their team representatives, who would then deliver the completed surveys to the principle investigators. While providing this option to the key contacts increased their trust in the research team and enhanced the level of engagement with and commitment to the research process, by relinquishing control of the survey distribution process the research team lost data that may otherwise have been available if the distribution process was administered solely by the principal investigators. There were also risks present for the patient/survivor participants. They gave of their time and energy that they may have experienced as compromised by disease and or treatment. They also consented to engage in a process that could stimulate negative memories and worries. As both of these risks were anticipated by the researchers, an on-site counselor was provided as well as a dedicated quite space for the duration of the workshop. Furthermore, the workshop was held in a conference room at the participants’ hotel, thus allowing for the opportunity to rest in their rooms as needed. The incorporation of patient/survivor voices and perspectives into the research process at all stages including instrument review, selection and development is critical to enhancing the validity and utility of cancer measures however the actual personal costs to the patient/survivors that provide their expertise has not yet been adequately assessed.

Implications While PR with cancer patient populations is still underutilized, the experience of the Survivor Dragon Boat Study provides further evidence of the potential of PR to increase responsiveness to patient needs and to improve the validity and relevance of instruments within cancer research, particularly in the realm of psychosocial research. When information is shared and power dynamics between researchers and consultants and advisory groups are balanced, participatory methods can result in significant methodological advances. PR has numerous unique advantages in knowledge creation and adds an important methodological tool for psychosocial cancer research. PR provides access to direct experience not

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via the data collection process alone, but also in the fundamental aspects of research question formation, research design, and instrument development and/or selection. By ensuring clarity of the items on psychometric instruments for a specific population, we can reduce the degree of error in survey responses by increasing the validity and reliability of the instrumentation. By attending to relevance, that is, asking how meaningful the research questions and survey items are to a specific research population, PR is a methodological vehicle to increase completion rates and advance knowledge relevant to policy and practice. By considering the question of comfort, researchers can attend to the issue of research burden and distress in relation to instrument completion. Participatory research requires additional time, resources, care and attention to the challenges of the process [5, 36]. Utilizing a participatory approach for collaborative research with a cancer population also involves unique considerations of ethics and care. Researchers are required to be conscious of the research burden, not only on research participants, but also on survivor/patient research consultants and advisory group members. Special attention is required for adequate rest, nutrition, debriefing, and emotional support throughout the research process. We recommend that beyond the basic PR principles of respect, power sharing, and reciprocity, that researchers working with cancer populations include in their research budgets resources to attend to issues and activities of care and comfort. In addition, we recommend that researchers incorporate social networking elements as an extended ethical dimension of participatory cancer research, and honoraria appropriate to the critical expert knowledge that patient/survivor consultants provide to research.

Conclusions We have described a comprehensive community-based PR research approach with an identified breast cancer population in Ontario, Canada. We shared our learning from a mixed method study in which qualitative findings, with 56 participant interviews, were utilized to identify key domains of interest to be investigated through a quantitative survey of 405 survivor dragon boaters, with an 81 % response rate. An advisory group of patient/survivors (five participants) was active through all phase of the research from grant development to dissemination of study findings. The PR approach was used to refine research questions, review and revise interview guides, and promote active community outreach to the study population. The PR approach also extended from the qualitative phase of the research into the quantitative phase. A larger group of patient/survivors (13 participants) were active in informing the development of a

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provincial survey instrument that included four psychometric measures that were selected for their clarity, comfort, and relevance, specific to the lived knowledge and experience of survivor dragon boaters. The development of the survivor dragon boat study and the survey package was carried out in the spirit of collaboration and learning from the population under study. We illustrated how increased clarity, comfort, and relevance from a patient/survivor perspective when developing or selecting research instruments relates to increased study rigor and ethics. The extensive PR approach was an effective means of honoring the sensitivities, challenges, and patient/survivor perspectives within the field of psychosocial cancer research. The breast cancer survivor dragon boaters participants were pivotal in guiding us to (a) refine the qualitative interview guides, (b) confirm our qualitative findings upon which a quantitative survey was constructed, (c) select the appropriate instruments and processes for a provincial survey, and (d) build relations of trust within the survivor dragon boat community across Ontario which facilitated recruitment and contributed to a high survey response rate. We believe such patient–researcher collaboration was a valuable and unique method of knowledge acquisition and contributed to enhancing ethics, rigor, and patient relevance of the research process and findings. Our experience in working with members of the cancer patient/survivor population on this study supports the findings of other researchers that patient participation in research design and instrument development contributes to higher completion rates of surveys, enhances design and conduct of the research while increasing the relevance of findings to the population under study [37–40]. Patient/survivor research consultants from the cancer population provided critical information on the selection, wording, and ordering of items and instruments. Sensitivity to the experience and needs of cancer patients within the research process is a strategy to increase completion rates, to reduce missing, data and increase the validity and reliability of the data, while enhancing ethical research practices with a vulnerable research population. Significantly, PR is not only a means by which the rigor of research generally can be enhanced but also a practical means by which we can increase the ethics of cancer research. In summary, our participatory process with a breast cancer patient/survivor population indicates that PR is an important methodological vehicle to improve the quality of psychosocial cancer research by increasing the validity of qualitative and quantitative research instruments, ethics in cancer research, recruitment processes, response rates, and dissemination. Further research is required to evaluate the costs and benefits to people living with cancer of their varied and active roles in participatory research.

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