RESEARCH ARTICLE For reprint orders, please contact: [email protected]

Engaging hard-to-reach patients in patient-centered outcomes research Aims: This study aimed to identify methods to engage hard-to-reach patients in the research process. Materials & methods: With funding from the PatientCentered Outcomes Research Institute (Washington, DC, USA), the University of Maryland (MD, USA) conducted 20 focus groups and one individual interview. The sample consisted of six groups of hard-to-reach patients, two groups of healthcare providers who work with hard-to-reach patients and two groups of surrogates of hard-to-reach patients. Results & conclusion: In order to make patient-centered outcomes research more meaningful to patients and their caregivers, patient-centered outcomes research should be conducted with a focus on building and maintaining trust, which is achieved via pre-engagement with communities and continuous engagement of study participants and their communities. KEYWORDS: African–American n comparative effectiveness research n minority n patient-centered outcomes research

Karen S Kauffman1, Susan dosReis2, Melissa Ross2, Beth Barnet3, Eberechukwu Onukwugha2 & C Daniel Mullins*2 University of Maryland School of Nursing, 655 West Lombard Street, Baltimore, MD 21201, USA 2 University of Maryland School of Pharmacy, 220 Arch Street, Baltimore, MD 21201, USA 3 University of Maryland School of Medicine, 29 South Paca Street, Baltimore, MD 21201, USA *Author for correspondence: Tel.: +1 410 706 0879 Fax: +1 410 706 5394 [email protected] 1

One of the most significant accomplishments of the Patient Protection and Affordable Care Act of 2010 was the establishment of the Patient-Centered Outcomes Research Institute (PCORI) [101]. PCORI’s missions are to assist patients, providers and the public in making informed health decisions, and to improve healthcare delivery and patient outcomes. While comparative effectiveness research (CER) is designed to reflect broad patient populations, researchers often do not successfully engage certain patient populations in the research process. Potential participants who are often excluded from research are sometimes referred to as ‘hard-to-reach’. These individuals are hard to reach as they are not often in settings and situations where research recruitment typically takes place. Eliciting the perspectives of diverse populations on health-related questions, not only from those who are convenient to recruit, is integral to the success of patient-centered outcomes research (PCOR). The primary goal of PCOR is to improve the health of all citizens by providing patients, their advocates and caregivers, healthcare professionals, policy-makers, payers, the government and other health agencies with evidence-based information that they can use to make informed healthcare decisions [1,2]. In reality, PCOR symbolizes a new era in which the most important stakeholder, the patient, is accorded a key role in research [3]. The concepts of patient and community involvement in research and clinical practice are not new. Community-oriented primary care, a concept introduced in the 1960s but not widely adopted, is a model in which primary care is provided to a defined community based on a health-needs assessment and the integration of public health practice [4]. Patient-centered medicine, a refocusing of medicine’s regard for the patient’s point of view, was evaluated by Laine and Davidoff [5]. Communitybased participatory research (CBPR) was being conducted in the early 1990s [6–9]; however, a 2006 Agency for Healthcare Research and Quality assessment found that CBPR had not been utilized to its full potential [10]. In addition, uncertainty in clinical, healthcare and health policy decision-making remains ubiquitous owing to large gaps in high-quality patient-centered medical evidence [11–13].

10.2217/CER.13.11 © 2013 Future Medicine Ltd

2(3), 313–324 (2013)

part of

ISSN 2042-6305

313

ReseArch article  

Kauffman, dosReis, Ross, Barnet, Onukwugha & Mullins

In November of 2011, PCORI awarded the University of Maryland (MD, USA) researchers a contract to conduct a study to identify methods for including hard-to-reach patients in research design, implementation and dissemination. The University of Maryland’s draft final report was sent to PCORI on 1  March 2012 so that the PCORI Methodology Committee could use the information as input for the PCORI’s report to Congress in May 2012. A private research firm, Westat (MD, USA), was subcontracted to assist in conducting focus groups. This research was guided by the questions outlined in the PCORI Patient Centeredness Working Group’s Request for Proposal. These questions, in their original form as stated in the request for proposals, are: ■■ What practical methods are effective in engaging patients and their surrogates in: identifying and prioritizing research questions; identifying outcomes that are meaningful to them; and identifying comparators/interventions that are meaningful to them? ■■

What methods are effective in ensuring that the full spectrum of patients is represented, including hard-to-reach and under-represented patients?

■■

What methods are effective in ensuring that informants understand the pertinent clinical and research issues (e.g., framing risk–benefit tradeoffs) so that results can meaningfully inform decision-making?

■■ Study procedures

This project had two phases of research. The goal of Phase I was to review the questions from PCORI and refine them as necessary for appropriate translation in a hard-to-reach population. Phase II involved the conduct and analysis of data generated from FG/Is with hard-to-reach patients and their care providers, including clini­cians (nurses and physicians), caregivers and parents. ■■ Phase I Stakeholder recruitment

What data are available that identify other factors and issues that are important to patients (or surrogates) that need to be considered? The findings described in this article are intended to guide researchers in developing methods to ensure that future CER and PCOR is meaningful to a broad array of participants, including those who are hard to reach. All study procedures were reviewed and approved by the University of Maryland institutional review board (protocols HP-00051288 and HP-00051504).

Nine key stakeholders, all of whom had experience working with hard-to-reach patients and/or conducting/developing PCOR methodologies, were invited to participate in Phase I. They were asked to assist with rephrasing the PCORI questions so that they would be meaningful to hardto-reach people, but still maintain the essence of the original questions. Five out of these nine stakeholders continued to serve as advisors and provided feedback throughout the research process to assure that our interpretation of findings was culturally relevant. Three assisted with identification of participants or communities for Phase II FG/Is.

Study participants & methods

Focus group procedures

The target population for this study was hardto-reach individuals. People were considered hard to reach if they belonged to a group that does not typically participate in research due to cultural or socioeconomic barriers, or owing to physical or cognitive impairment. An initial meeting with nine key stakeholders was held prior to conducting 20 focus groups and one individual

During a half-day meeting, the key ­stakeholders reviewed the questions posed by PCORI and offered expert input and feedback. The researchers then summarized the key points of the review and presented the recap to the group to ensure that the summary accurately reflected the discussion. The results of this meeting were used by the research team as a framework for the FG/I guide

■■

314

interview (FG/Is) with hard-to-reach patients or their care providers. The FG/Is included: six groups of hard-to-reach patients (low socioeconomic status African–American and Spanish/ English-speaking patients, African–Americans from faith-based organizations) and patients with mobility, vision and hearing impairments); two groups of healthcare providers (physicians who treat hard-to-reach patients and nurses who treat hard-to-reach patients); and two groups of surrogates (parents of children with mental health diagnoses and caregivers of patients with cognitive impairments). The individual interview was conducted with a caregiver who was unable to attend a focus group; the nurse was asked the same questions as were those in the focus group.

J. Compar. Effect. Res. (2013) 2(3)

future science group

Engaging hard-to-reach patients in patient-centered outcomes research 

that would be used in Phase II. FG/I guides are available from the authors, as well as online in the final report [102]. No further analysis of this focus group was done. ■■ Phase II Participant recruitment

Participants were recruited from eight key Baltimore-based organizations: Bon Secours Baltimore Health System; the practice-based research network comprised of community health centers; the Association of Black Cardiologists; community- or faith-based organizations (e.g., Mount Lebanon Baptist Church); the Alzheimer’s Association Greater Maryland Chapter; Weinberg Senior Living; the Maryland Coalition of Families for Children’s Mental Health; and the University of Maryland School of Nursing. Bon Secours Baltimore Health System assisted with patient recruitment related to racial/ethnic diversity and those with low socioeconomic status. To further expand the diversity within each of the hard-to-reach categories, recruitment focused on varying participant ages and geographic locales, as well as both males and females. In addition, for the subpopulations that were not defined by race/ethnicity, we purposefully recruited ­A frican–American participants. FG/I procedures

FG/Is were scheduled at varying times of the day and on different days of the week, including weekends, to accommodate each group’s schedule and preference. Either lunch, snacks or a light dinner was provided for the participants and each person received a US$25 gift card for a local chain pharmacy as compensation for their time. Members of the University of Maryland research team (KS  Kauffman and S  dosReis) and Westat (L Jansky, J Huang) with experience in facilitating focus groups led the focus groups and other members of the research team (M Ross, P Lipman) were note-takers. Written consent was obtained from all participants before beginning the focus-group discussion. From initial consent to conclusion of the discussion, the focus groups lasted between 1 and 2 h. During the discussion, the interviewers/moderators engaged all participants in a manner that was inclusive and nonjudgmental to encourage expression of diverse viewpoints. Notes were collected in real time using flip charts and a laptop. The discussions were audio recorded, with consent of ­participants, and transcribed.

future science group

ReseArch article

Analysis of Phase II FG/I

Three members of the research team with expertise in the analysis of qualitative data (Kauffman, dosReis and Onukwugha) independently reviewed and coded a set of seven to eight FG/I transcripts to establish a preliminary set of codes. The defining characteristics of each code were discussed among the group of researchers and codes were refined, expanded or collapsed based on the group consensus. Using the refined set of codes, each coder reread the interviews to ensure that all relevant information was identified. The coded data were again discussed among the group, and codes were further refined as needed. At this stage, a coding manual was developed. The coders then moved onto the remaining interviews using the established codes. Any new codes that emerged were discussed among the group and added to the coding manual. The prior set of interviews were again reviewed to ensure that all information related to the existing and newly identified codes had been captured. This process continued until there was 100% agreement in definitions and coded data. The coding manual evolved throughout the process until saturation was achieved – that is, saturation occurred when no new codes were identified in each subsequent transcript. The codes were categorized according to the three overarching research questions: how to engage hard-to-reach patients? How to ensure informants’ understanding? What data are available to identify other factors important in the process of engaging hard-to-reach patients? The three coders met over 3 half-days to discuss the coding and categorizing and to resolve differences. Major themes emerged from the sorting of codes in each of the three categories, which were then compared and contrasted to assure an exhaustive list without repetition. Additional contextual and procedural data that were not directly related to the research questions, but were thought to hold potentially important information to inform key recommendations for engaging patients in PCOR, were also analyzed for themes. The transcribed interviews were uploaded into NVivo 2.0 using the codes identified and agreed upon by the three researchers (Kauffman, dosReis and Onukwugha). A coding summary table was generated to visually display the ­distribution of codes within and across FG/Is.

www.futuremedicine.com

315

ReseArch article  

Kauffman, dosReis, Ross, Barnet, Onukwugha & Mullins

Results ■■ Characteristics of the Phase II FG/I participants

The race/ethnicity and gender distributions of participants for each of the ten categories of hardto-reach patients who were interviewed for this study are displayed in Table 1. The race/ethnicity distribution of the participants across the FG/Is is consistent with the population census for the Baltimore area. Qualitative analysis of the FG/I transcripts was organized by each of the key PCORI questions. The main thematic concepts that emerged are discussed below, and the distribution of the themes across the FG/Is is illustrated in Table 2.

the block and ‘the man in the trench’, individuals who live in the same community as the participants and ‘look just like them’. Groups of people include healthcare professionals, employers, support group members, law enforcement and justice officers, and public service workers. Associations of people include community groups, social organizations, such as sororities and fraternities, and federations such as the National Federation of the Blind. In rural ­settings, networks most often ­comprised families. Meeting in public places and private spaces

Practical methods for engaging hard-to-reach patients include partnering with people in formal and informal community leadership positions to provide entrée to meeting places frequented by hard-to-reach people. In addition to partnering with community leaders, participants thought that using media would spread the word about opportunities for individuals to learn about and participate in research. Each of these methods is discussed in further detail below.

Participants identified a wide range of public places and private spaces that hard-to-reach individuals frequent. Public spaces open to everyone include street corners and local businesses such as laundromats, hair salons, banks and grocery stores. Community-specific spaces where community members receive services include centers for recreation, relocation, social services, and senior-citizen activities. Semiprivate places include emergency rooms and community clinics where hard-to-reach people who are underinsured or uninsured frequently seek healthcare, and places of worship where people gather to share common ground and beliefs. Private spaces include housing such as long-term care facilities, shelters, jails and prisons.

Partnering with people

Using media

Participants identified a wide range of individuals, groups and associations with whom to partner for PCOR. The list includes the ‘mayor’ of

Types of media, a means for mass communication and social marketing, have varying ranges of reach. Narrow-reach media are culture-specific,

■■ Practical methods for engaging patients & ensuring representation of hard-to-reach patients

Table 1. Race/ethnicity and gender of participants in ten categories of focus groups/interviews. Focus group/interview category

Af–Am (F), n

Af–Am

6

Faith-based

23

Visually impaired

1

Hearing impaired

1

Mobility impaired

9

Bilingual (Spanish speaking)

Hisp (F), n

NHWh (F), n

Other (F), n

40

Caregivers

9

Parents

15

Nurses

4

Physicians

8

Grand total

76

Hisp (M), n

NHWh (M), n

Other (M), n

6

Total 12 23

1

2

3

9

1

7

1

11

Patient total

Af–Am (M), n

12

12

18

0

1

6

9

6

1

8

4

15

2

19 18

9

1

95

3

12

2

17

14

1

19

1

1

5

33

2

19

6

1

1

17

10

2

160

Af–Am: African–American; F: Female; Hisp: Hispanic; M: Male; NHWh: Non-Hispanic white.

316

J. Compar. Effect. Res. (2013) 2(3)

future science group

Engaging hard-to-reach patients in patient-centered outcomes research 

ReseArch article

Table 2. Major themes discussed in focus groups. Interview

A

B

C

D

E

F

G

H

I

J

n

1

1

1

1

1

1

1

10

1

1

1

1

1

1

1

1

1

1

10

1

1

1

1

1

1

9

1

1

1

10

1

1

9

1

1

9

1

8

Question 1 topic: methods of engaging Places to go

1

1

1

Special events

1

1

1

Organized groups

1

1

1

Media and advertising

1

1

1

Individuals

1

1

1

1

1

1

1

Building trust/being there

1

1

1

1

1

1

1

Building trust/relating

1

1

1

1

1

1

1

Building trust/pre-engagement

1

1

1

1

1

1

1

Building trust/communicating

1

1

1

1

1

1

1

1

1

9

Incentives

1

1

1

1

1

1

1

1

1

9

Instrumentation

1

1

1

1

1

1

1

1

1

Uncategorized

1

1

1

1

1

Literacy and comprehension

1

1

1

1

1

1

Consent process

1

1

1

1

1

Information gathering

1

1

1

1

Disclosure

1

1

1

1

1

1

1

1

1

7

1

10

1

1

9

1

1

9

1

6

Question 2 topic: understanding

Uncategorized

1

4 1

6

1

4

Question 3 topic: identifying important factors Health and medical

1

1

Socioeconomic indicators

1

1

1

1

1

Community resources

1

1

1

1

1

Difficult life situations

1

1

Social networks

1

Social media

1

Uncategorized

1

Medical professional health facility

1

1 1

1

1

1

1

1

1

1

1

1

1

1

1 1

5

1

1

8

1

1

7

1 1

1

5 1

7 6

1

1

1

4

1

1

1

5

A: Caregivers of individuals with cognitive impairments; B: Nurses who treat hard-to-reach individuals; C: Parents of children with cognitive impairments; D: Physicians who treat hard-to-reach individuals; E: Low socioeconomic status (Spanish/English patients); F: African–Americans from faith-based organizations; G: Individuals with hearing impairments; H: Low socioeconomic status (African–American patients); I: Individuals with mobility impairments; J: Individuals with vision impairments; n: Total number of groups that brought up the theme.

targeting local or regional communities, including billboards on the side of buses and hospital buildings, flyers in utility bills and other mailings, and local television and radio stations. Media-reaching groups, beyond the local and regional communities, include national television and radio, as well as clips and trailers before the start of films. Media with the widest range include online ­bulletin boards, list serves, Facebook® and Twitter®.

of the research include the researcher knowing participants’ level of literacy and comprehension of what is being said to them, using culturally competent means to gather information and obtain consent, and providing full disclosure of the intent of the research as well as that of the researcher. The spectrum of issues that were raised by the study participants is described briefly below for each topic.

■■ Methods to ensure patients understand pertinent issues

Literacy & comprehension

Methods to ensure that participants fully understand the pertinent clinical and process issues

future science group

The concerns about a study participant’s ability to understand the research reflect the fundamentals of using plain language as opposed to

www.futuremedicine.com

317

ReseArch article  

Kauffman, dosReis, Ross, Barnet, Onukwugha & Mullins

‘medical language’. As there is an element of shame associated with illiteracy, one must be careful that an individual is not covering up their illiteracy by simply agreeing with the process. The best way to overcome problems with literacy and comprehension is to be straight­ forward and specific, and ask participants if they understand and to repeat things back in their own words and phrases – that is, ‘teach back’. Another strategy is to offer a class or course for individuals to understand the research. Gathering information

Using the appropriate terminology when gathering information is important since ‘research’ is an emotionally charged word. Asking open-ended questions creates opportunities to hear study participants’ questions and concerns in their own words and avoids imposing the researchers’ preconceived ideas about what is important to the study participants. Partnering with community members, such as community health volunteers, assures cultural competence in responding to the questions and concerns and increases the likelihood that participants will understand the ­information that is being conveyed. Consent process

Participants noted that the institutional review board consent forms use scientific jargon that most people do not understand and fine print that people do not or cannot always see. The long consent forms “make patients nervous about what they are getting into” because the “risks are buried on page 8 of a 17-page form.” A way to address the long, cumbersome consent forms is to “culturally match the ‘consenter’ to the person” or have individuals consent in “their own words.” During this process, it is important for the researcher to keep in mind that “people may just need what you are offering … they may not care about risks and benefits,” and that full understanding may not be achieved the first time. Individuals may need time to think about the study, talk with others and then come back with more questions to reach a fully informed decision. Disclosure

Participants reflected on the process of providing full disclosure about the research, a process that is intended to avoid study participants from feeling as if they are ‘laboratory rats’. In this process, not only “what you say, but who says it” is essential to the participants. Full disclosure involves total

318

J. Compar. Effect. Res. (2013) 2(3)

honesty about the researcher’s expectations of the study participants, a clear explanation of risks and benefits of participation and “what is in it for the researcher”. It is as important to let individuals know exactly what is involved as it is to tell them why the investigator is doing the research. ■■ Data to identify issues important to hard-to-reach patients

Data that identify other factors and issues important to hard-to-reach people include particular information about their community resources and difficult life situations. Composite information includes health and medical records, socioeconomic indicators, and social media and networks. The following text provides additional participant feedback regarding these topics. Community resources

A fundamental understanding of “how you define community” and the process of going ‘door-to-door’ leads to successful collection of information about local resources. Knowing who relies on public transportation, why they rely on it and where they go indicates the types and availability of community resources. Other indicators of community resources include the availability and proximity of parks and recreation areas, community centers, supermarkets, corner stores, fast food restaurants and the availability of fresh fruits and vegetables versus ‘junk food’. Difficult life situations

Many people who are hard to reach have life experiences that place them at risk for judgment, stigmatization and marginalization from their families, friends and communities. Examples of available data to identify them include police records and frequent emergency service calls to fire departments and ambulance services. Crime rates identify communities at risk. Health & medical records

The health of hard-to-reach people can be gleaned from records from medical offices, pharmacies and community-based social services. Such places include emergency rooms where uninsured and underinsured people gain access to care, schools where data on the use of school lunch programs indicate the health and lifestyle of the child as well as the family (since one can “follow a fat kid home and find a fat parent”), in addition to ‘well baby clinics’ and the Special Supplemental ­Nutrition Program for Women, Infants and Children.

future science group

Engaging hard-to-reach patients in patient-centered outcomes research 

Socioeconomic indicators

Traditional census-based data including demographics and socioeconomic indicators are available to identify other factors important to people; however, study participants also reflected on the importance of knowing “where people are spending their money” or whether they need help to pay the bills. Information regarding financial assistance needs can be found by searching utility company records for “turn off notices,” and records of energy-assistance p­rograms and ­disability compensation. Social networks & social media

Social networks inf luence people in many aspects of their lives, including healthcare decisions. Local networks include support groups, friends, neighbors and translators for those for whom English is not their first language. Social media that have a wider reach include radio and community newsletters. Since social media can promote ‘negative imagery’ about people and communities, understanding how such media affect people is a ­potentially important source of data. ■■ Themes for guiding future PCOR studies

Ten additional themes emerged during the interviews that address contextual and procedural issues for carrying out a PCOR study; they provide guidance for meaningfully engaging study participants (patients, community partners and surrogates) in PCOR. These additional themes address how and where to engage study participants and how to keep them engaged in PCOR (Box 1). Bring PCOR to the communities where people live

Frequently, researchers require participants to come to them, which often results in including only the most motivated patients. Optimal

ReseArch article

engagement can only occur when the research is conducted where participants live or congregate, such as recreation centers, grocery stores and neighborhood blocks. This process will ­probably increase diversity and inclusivity. Use a period of ‘pre-engagement’ when r­ecruiting research participants & partners

Pre-engagement means spending time with study participants and community partners more than once prior to having them sign an institutional review board consent. This time allows the researcher to understand the environmental context in which people live and what barriers/facilitators may impact the research study. It allows study participants time to ask questions, weigh the benefits versus risks of participation more fully and discuss issues and concerns with family and friends. This process facilitates cultural sensitivity and competence among the researchers. Involve the full spectrum of people that will be affected by the research, including hard-to-reach patients

Government funding agencies typically require studies to be conducted with priority populations; however, since the recruitment is often limited to a catchment area for convenience, important subpopulations are left out. In these cases, generalizability may be adversely affected. Build & maintain trust for active study participant engagement

Establishing mutual trust between the researcher and the study participants builds bridges for open and enduring engagement, and is essential in producing valid and reliable PCOR results. Establishing trust takes time and requires deliberate and ongoing communication, but can easily be lost if a study participant feels misled

Box 1. Key recommendations for meaningfully engaging patients in patient-centered outcomes research. Bring patient-centered outcomes research to communities where people live Use a period of ‘pre-engagement’ when recruiting research participants and partners ■■ Involve the full spectrum of people that will be affected by the research, including hard-to-reach patients ■■ Build and maintain trust for active patient engagement ■■ Provide education on what is meant by the term ‘research’ ■■ Realize that people may not be willing to openly disclose medical information ■■ Keep people up-to-date on what is going on with the research ■■ Provide a lay summary of study findings at an end-of-study celebration ■■ Make a sincere effort to ‘give back’ to the community ■■ Recognize that people make healthcare choices and participate in research based upon who they are as individual persons, not just as patients ■■ ■■

future science group

www.futuremedicine.com

319

ReseArch article  

Kauffman, dosReis, Ross, Barnet, Onukwugha & Mullins

about the nature of the research. The process of building and maintaining trust is fundamental to successfully carrying out PCOR. Provide education on what is meant by the term ‘research’

The term ‘research’ carries a negative connotation in some communities. This results in distrust and limited participation from traditionally under-represented groups. Educating study participants about research must begin during pre-engagement with the community and continue throughout the study, and include information on research in general and, in particular, the PCOR project. Understanding what research is and how results are used will facilitate study participants having positive experiences. It is also important for participants to understand that participating in a PCOR study is not an opportunity to receive care or obtain i­nformation about the conditions or treatments. Realize that people may not be willing to openly disclose medical information

Some people are not willing to disclose medical information from fear of being ostracized from their community, or due to having cultural norms in which personal information should be ‘kept in the family’. Developing methods for assuring patient confidentiality and explaining to study participants how personal information will be kept confidential will promote ­transparency and trust. Keep people up-to-date with what is going on with the research

In order to keep study participants fully engaged, they need to be kept fully informed throughout the research process. Often, large gaps in time occur between data collection points when study participants do not have contact with the researcher. During these gaps, participants can lose interest in following the research protocol or question the value of the research itself, jeopardizing the study’s scientific integrity. In addition, providing feedback to participants in ‘real-time’ engenders trust and goodwill that will probably result in willing engagement in future research studies. During sessions when updates are presented, participants should be actively engaged so that participants can provide their interpretation of results. Alternatively, the updates can provide the opportunity to seek participants’ feedback on draft documents summarizing the research

320

J. Compar. Effect. Res. (2013) 2(3)

team’s consensus regarding the ­interpretation and meaning of study results. Provide a lay summary of study findings at an end-of-study celebration

Providing a summary of study findings at the end of the study keeps participants informed and engaged. It recognizes their significant contributions to the success of the study, and it also presents an opportunity for them to participate in a formative evaluation of the research process – that is, what worked well and what could be improved in the next study. Recognizing participants as a partner in the design of future studies demonstrates respect for their contributions to science and contributes to the successful design of future studies. Make a sincere effort to ‘give back’ to the ­community

Participants would like researchers to ‘give back’ to their communities in ways that expand beyond providing a summary of study findings at the end of the study. Returning to the community just to say hello or to participate in a community event that is unrelated to the investigator’s research project shows a genuine ­commitment to them and their communities. Recognize that people make healthcare choices & participate in research based upon who they are as individual persons, not just as patients

Using the term ‘patient’ implies a health condition that requires management by a healthcare provider, who often only knows the person within the context of the medical model. It often implies a top–down relationship of a provider with the individual who is expected to ‘comply’ and ‘adhere’ to a treatment protocol. In research, study participants who are called ‘patients’ often feel they are ‘laboratory rats’ who have limited engagement with the researcher and are narrowly viewed as data contributors for the study. Viewing participants as ‘people’ rather than ‘patients’ involves recognizing their social and physical environments and the broader context in which people make decisions to participate in research and reflects respect for their significant contributions to the research process. Discussion

The signing of the Patient Protection and Affordable Care Act in 2010 stimulated a new way of doing CER, aimed at improving healthcare delivery and patient outcomes to reduce health

future science group

Engaging hard-to-reach patients in patient-centered outcomes research 

disparities [101]. Previously, the researcher took charge of every step of the research process: conceptualizing CER questions; choosing comparator interventions; and determining processes for study participant selection and recruitment, data collection and dissemination of results. The targeted beneficiary of the research, the patient, had little voice in the process and, most often, never knew the researcher or even the results of the study. Furthermore, targeting hard-to-reach patients was not necessarily emphasized. With the advent of PCORI, the role of patients as study participants has begun to evolve into one of partnering with the researcher, and engaging hard-to-reach patients in the PCOR process has become an over-riding goal. This article presented recommendations to facilitate success in PCOR based on the questions posed by PCORI in the contract; however, what emerged as the essential PCOR prerequisite is the building and maintaining of trust between the researcher and the patients who participate in the study, as well as the community to which they belong. The process of gaining, building and maintaining mutual trust with the group under study is a common practice called ‘getting in’ among qualitative researchers, especially ethnographers, and is a fundamental principle in CBPR [13,14]. It takes time, deliberation, commitment and resources, most of which are incorporated into the funding proposal. Most often, the process targets a discrete and well-defined group or community with social characteristics different from those of the researcher. It also requires that both clinical and qualitative researchers partner with the community so that community members are active participants in the design and execution of PCOR, and are not just research subjects. Incorporating a new set of processes into CER to achieve the PCORI goal to engage hard-to-reach patient populations will require a new way of doing things (e.g., including best practices of qualitative research, CBPR, population-focused research and clinical trials); carrying out methodological research to identify ways to successfully engage and retain study participants who are least likely to participate in CER; preparing and retraining researchers to work simultaneously with other researchers from different paradigms; and funding support for research that is more complex and may require more resources and time to complete. The research may also take longer to ‘begin’ if community pre-engagement is incorporated. The pre-engagement process introduces a phase

future science group

ReseArch article

that many researchers have neither systematically implemented in their research nor incorporated into grant proposals. The successful implementation of the pre-engagement phase will require ‘time and materials’ support from researchers, as well as financial support from sponsors of research. Productive discussions regarding the responsibilities of researchers versus sponsors in financially supporting the pre-engagement phase will continue and will be important for the successful and sustained incorporation of pre-engagement into PCOR. The topics discussed do not represent an exhaustive action list; however, the discussion provides recommendations based on our findings that are critical to consider in moving forward the PCORI agenda to reduce health disparities through the conduct of CER [15]. Limitations

Except for nurse practitioners working in the rural eastern shore of Maryland, all other focus group participants in both Phase I and II were from the greater Baltimore area. As in most qualitative research, findings of this study may not be transferable to other hard-to-reach populations and communities beyond Baltimore. However, we believe that the findings will provide valuable guidance for conducting PCOR in other similar urban environments. Conclusion

When conducting PCOR in hard-to-reach populations, CBPR and ethnographic principles about building and maintaining trust should be incorporated into CER to contribute to the reliability and validity of study findings. Pre-engagement is necessary to assure that community partners and patients understand the research and can trust the researchers. Even within minority populations, there are some individuals who are easier to recruit than others. Pre-engagement will help researchers develop cultural competency with hard-to-reach populations. Trust is more likely to be maintained through continuous engagement, even after a research study is complete. Future perspective

Within the next decade, minorities will outnumber non-Hispanic whites in the USA; therefore, more evidence regarding minorities and hardto-reach patients will become even more critical for improving the health of the nation. Enga­ ging hard-to-reach patients in CER and PCOR studies will increase broader representation of

www.futuremedicine.com

321

ReseArch article  

Kauffman, dosReis, Ross, Barnet, Onukwugha & Mullins

Executive summary Background ■■ One of the most significant accomplishments of the Affordable Care Act of 2012 was the establishment of the Patient-Centered Outcomes Research Institute (PCORI). ■■ The primary goal of patient-centered outcomes research (PCOR) is to improve the health of all citizens by providing patients, their advocates and caregivers, healthcare professionals, policy-makers, payers, the government and other health agencies with evidence-based information to make informed healthcare decisions. ■■ The University of Maryland (MD, USA) researchers were awarded a PCORI contract to identify methods for including hard-to-reach patients in research. Study participants & methods ■■ Twenty focus groups and one interview were conducted with six groups of hard-to-reach patients (low socioeconomic status African–American and Spanish/English-speaking patients, African–Americans from faith-based organizations) and patients with mobility, vision and hearing impairments); two groups of healthcare providers (physicians who treat hard-to-reach patients and nurses who treat hard-to-reach patients); and two groups of surrogates (parents of children with cognitive impairments and caregivers of patients with cognitive impairments). ■■ The study had two qualitative research phases: in Phase I, key stakeholders with experience in working with hard-to-reach patients reviewed the questions from PCORI and refined them as necessary to prepare the interview guide; and in Phase II, focus groups and one interview were conducted with hard-to-reach patients. ■■ The interviews were transcribed, then analyzed into codes and categorized according to the three overarching research questions. Results ■■ Practical methods for engaging patients and ensuring representation of hard-to-reach patients include partnering with people in formal and informal community leadership positions to provide entrée to meeting in places frequented by hard-to-reach people. ■■ Methods to ensure that participants fully understand the pertinent clinical and process issues of the research include the researcher knowing participants’ level of literacy and comprehension of what is being said to them, using culturally competent means to gather information and obtain consent, and providing full disclosure of the intent of the research, as well as that of the researcher. ■■ Data that identify other factors and issues important to hard-to-reach people include particular information about their community resources and difficult life situations. Composite information includes health and medical records, socioeconomic indicators, and social media and networks. ■■ Ten additional themes emerged during the interviews that address contextual and procedural issues for carrying out a PCOR study; they provide guidance for meaningfully engaging study participants (patients, community partners and surrogates) in PCOR. These additional themes address how (i.e., pre-engagement) and where (i.e., within communities) to engage study participants and how to keep them engaged in PCOR (i.e., give back to the community and keep people up to date). Discussion ■■ This article presented recommendations to facilitate success in PCOR, based on the questions posed by PCORI in the contract; however, what emerged as the essential PCOR prerequisite is the building and maintaining of trust between the researcher and the patients who participate in the study, as well as the community to which they belong. ■■ Incorporating a new set of processes into comparative effectiveness research (CER) that will achieve the PCORI goal of including hard-to-reach patient populations will require a new way of doing things. Examples include combining best practices of qualitative research, CBPR, population-focused research and clinical trials; conducting methodological research to identify ways to engage and retain study participants who are least likely to participate in CER; preparing and retraining researchers to work simultaneously with other researchers from different paradigms; and gaining funding to support research that is more complex, may require more resources and may take longer to complete. Limitations ■■ As in most qualitative research, findings of this study may not be transferable to other hard-to-reach populations and communities beyond Baltimore. Conclusion ■■ Pre-engagement is necessary to assure that community partners and patients understand the research and can trust the researchers. ■■ Even within minority populations, there are some individuals who are easier to recruit than others. ■■ Trust is more likely to be maintained through continuous engagement, even after a research study is complete. Future perspective ■■ More evidence regarding minorities and hard-to-reach patients will become even more critical for improving the health of the nation. ■■ By applying and refining “best practices” for engaging hard-to-reach patients in CER and PCOR, future studies will allow care providers to provide culturally competent and clinically appropriate care for all patients, including hard-to-reach patients.

322

J. Compar. Effect. Res. (2013) 2(3)

future science group

Engaging hard-to-reach patients in patient-centered outcomes research 

populations that were previously least likely to participate in research. Such studies alone, however, are insufficient to assure that populations and communities receive culturally competent and clinically appropriate care. Applying evidence to clinical practice requires knowledge and willingness by both providers and recipients of healthcare services. Additional methodo­logical research must include studies to identify best practices for incorporating evidence into clinical practice in order to achieve the goals of PCOR. Acknowledgements The authors gratefully acknowledge the assistance of L Frank from Patient-Centered Outcomes Research Institute; L Jansky, P Lipman and J Huang from Westat for their assistance with designing, conducting and summarizing the focus groups; J Vandigo, who provided support as a graduate student research assistant; A Laird and S Ross from Bon Secours Baltimore Health System; F Lance, C Johnson, D Mack and D Martin from Mt Lebanon Baptist Church; D Smith and A Williams from the Association of Black Cardiologists; S Alborn, Executive Director of Community Health Integrated Partnership (CHIP); others who helped to recruit participants; and most importantly the patients and care providers who ­participated in our interviews.

Papers of special note have been highlighted as: of interest n

1

n

2

n

Bonham AC, Solomon MZ. Moving comparative effectiveness research into practice: implementation science and the role of academic medicine. Health Aff. (Millwood) 29, 1901–1905 (2010). Provides a thorough explanation of the connection between comparative effectiveness research (CER) and implementation science. Aimed, in particular, toward academic medicine participants: administrators, physicians, scientists, nurses, pharmacists, other staff members and trainees, the authors promote interdisciplinary leadership to develop new models of healthcare – that is, to become ‘a comprehensive learning laboratory’ that remains focused on patient-centered outcomes. Lauer MS, Collins FS. Using science to improve the nation’s health system: NIH’s commitment to comparative effectiveness research. JAMA 303, 2182–2183 (2010). Brings to focus five important challenges for the NIH and the Agency for Healthcare

future science group

Disclaimer The views expressed in this article are those of the authors and do not necessarily reflect those of the Patient-Centered Outcomes Research Institute.

Financial & competing interests disclosure D Mullins received funding via a contract from the PatientCentered Outcomes Research Institute to conduct “Expert Stakeholder Interviews to Identify Evidence for Eliciting the Patient’s Perspective in Patient-Centered Outcomes Research”. This funding supported the research described in this article. The authors have no other relevant affiliations or financial involvement with any organization or entity with a financial interest in or financial conflict with the subject matter or materials discussed in the manuscript apart from those disclosed. No writing assistance was utilized in the production of this manuscript.

Ethical conduct of research The authors state that they have obtained appropriate insti­tutional review board approval or have followed the princi­ples outlined in the Declaration of Helsinki for all human or animal experimental investigations. In addition, for investi­gations involving human subjects, informed consent has been obtained from the participants involved. Research and Quality as they lead the nation forward in CER: continuing stakeholder involvement; the acceptance of new methodologies embedded in CER; the use of nonexperimental studies; the importance of implementation science as a tool to improve dissemination of findings; and the leveraging of multidisciplinary expertise in high-throughput technologies. In addition, the role of personalized medicine is reiterated as an essential part of the equation for improving healthcare.

References

3

n

ReseArch article

Conway PH. How the Recovery Act’s Federal Coordinating Council paved the way for the Patient-Centered Outcomes Research Institute. Health Aff. 29(11), 2091–2097 (2010). Explains the formation of the governing board for Patient-Centered Outcomes Research Institute, its responsibilities to stakeholders and its priorities for research. Based upon the prior work of the Federal Coordination Council, Patient-Centered Outcomes Research Institute has a ‘heads-up’ in what research has been accomplished and where gaps still exist. The article stresses that collaborative efforts are necessary across federal, state and local funding agencies in

www.futuremedicine.com

order for this newly formed, nongovernmental agency to meet the goals of coordinated CER. 4

Mullan F, Epstein L. Community-oriented primary care: new relevance in a changing world. Am. J. Public Health 92(11), 1748–1755 (2002).

5

Laine C, Davidoff F. Patient-centered medicine: a professional evolution. JAMA 275(2), 152–156 (1996).

6

Cornwall A, Jewkes R. What is participatory research? Soc. Sci. Med. 41(12), 1667–1676 (1995).

7

Israel BA, Schulz AJ, Parker EA, Becker AB. Review of community-based research: assessing partnership approaches to improve public health. Annu. Rev. Public Health 19, 173–202 (1998).

8

Viswanathan M, Ammerman A, Eng E et al. Community-Based Participatory Research: Assessing the Evidence. Evidence Report/ Technology Assessment No. 99 (Prepared by RTI–University of North Carolina) (2004).

9

Community-Based Participatory Research for Health. Minkler M, Wallerstein N (Eds). Jossey-Bass, San Francisco, CA, USA (2003).

323

ReseArch article   10

11

n

Kauffman, dosReis, Ross, Barnet, Onukwugha & Mullins

Evidence-based Practice Center under Contract No. 290–202–0016). AHRQ Publication 04-E022-2. Agency for Healthcare Research and Quality Rockville, MD, USA (2004). Mullins CD, Whicher D, Reese E, Tunis S. Generating evidence for comparative effectiveness research using more pragmatic randomized controlled trials. Pharmacoeconomics 28(10), 969–976 (2010). Postregulatory decision-makers should be engaged in the design and operationalization of pragmatic clinical trials, which are trials that occur in more real-world populations. This article describes the need to engage key stakeholders and generate evidence within the context of pragmatic trials in order to maximize the impact of health-improving interventions.

12 Mullins CD, Montgomery R, Tunis S.

treatments. Oncologist 15(Suppl. 1), 58–64 (2010). 13 Analyzing Social Settings (2nd Edition).

Lofland J, Lofland LH (Eds). Wadsworth, Belmont, CA, USA (1984). 14

15

n

Kauffman KS. The insider/outsider dilemma: field experience of a white researcher “getting in” a poor black community. Nursing Res. 43, 179–183 (1994). Mullins CD, Onukwugha E, Cooke JL, Hussain A, Baquet CR. The potential impact of comparative effectiveness research on the health of minority populations. Health Aff. 29(11), 2098–2104 (2010). CER has great potential to produce meaningful evidence that would support a reduction in health disparities among minority populations and other subgroups. The authors recommend methods such as

more informative subgroup analysis, increased recruitment of minorities and receiving input from community health workers to ensure research is relevant to the needs of minority patients.

■■ Websites 101 The United States Government Printing

Office. Patient Protection and Affordable Care Act. Public Law 111–148. www.gpo.gov/fdsys/pkg/PLAW-111publ148/ pdf/PLAW-111publ148.pdf (Accessed 24 September 2012) 102 Integrating patients’ voices in study design

elements with a focus on hard-to-reach populations (page 50–59). http://pcori.org/assets/pdfs/Integrating%20 Patients%20Voices.pdf (Accessed 24 September 2012)

Uncertainty in assessing value of oncology

324

J. Compar. Effect. Res. (2013) 2(3)

future science group