Patient and consumer engagement is now internationally recognized as an important component of clinical practice guideline development. This engagement recognizes that patients are experts, respects the rights of citizens in health policy development, empowers and informs consumers in healthcare decisions, and leads to the development of more patient-centered and trustworthy guidelines [1]. Patient and consumer engagement impacts key elements of recommendation development, including identifying whether the problem is a priority, determining if effects are meaningful, weighing risks and benefits, analyzing the impact of costs, and assessing acceptability and feasibility [2]. For these reasons, several organizations recommend or require that patients, patient representatives, or health consumers be engaged on guideline development panels alongside physicians and other professional stakeholders, including the Guidelines International Network (G-I-N) [3], the United Kingdom’s National Institute for Clinical Excellence [4], and the United States’ Institute of Medicine [5].

While there is now widespread acceptance of the value of engaging patients, scant evidence exists on whether this takes place or optimal methods by which to engage patients. A survey of guideline developers found that only 29% always involve consumers and 39% involve consumers “only if necessary” [6]. This may reflect known barriers to engaging patients and consumers in guideline development, including uncertainty of how to incorporate patient experiences into evidence-based guidelines [7], the resources needed to engage patients well [7, 8], participant feelings of isolation [8], difficulty understanding medical terminology and assessing research quality [3, 4, 7–9], recruitment difficulties [8], and inadequate training and support [4, 7, 9].

A 2006 Cochrane review found no trials investigating public involvement in guidelines and concluded that “little research has been done to find the best ways of involving consumers in healthcare decisions at the population level” [10]. There is a similar paucity of data when considering best practices for engaging patients in research settings (as opposed to guidelines) [11, 12]. Most suggestions for engaging patients and consumers stem from the practical experiences of guideline developers. These experiences suggest the importance of recruitment strategies [13], including at least two patient/consumer representatives [9], considering compensation [1], early engagement continuing throughout the guideline process [9], training [1, 4, 13, 14], support [1, 4, 9, 13], clear communication [14], clarity regarding the patient role [4], equitable treatment [14], managing group dynamics [1], skilled moderation [4], specifying guideline scope [4], making discussion of patient and caregiver issues a specific agenda item [4], and funding additional methods of soliciting patient views when needed [4].

No identified research identifies the views of patients and consumers not yet recruited to participate in guideline development. This is an important knowledge gap. While some guideline developers use experienced patients and consumers, such as those trained through Consumers United for Evidence-Based Healthcare, many developers recruit patients and patient representatives without prior training or experience. Research suggests that public awareness of guidelines is low and that individuals aware of guidelines have only a vague understanding of what they are and how they are developed [15]. Given this, and given that difficulty recruiting is one of the most frequently cited barriers to patient and public involvement in guidelines [8], investigating the views of potential participants has the potential to generate important guidance for developers seeking to engage guideline-naïve patient and consumer representatives.

Additionally, despite the above forays into highlighting practical mechanisms for patient and consumer engagement in guidelines, critical questions remain unanswered, such as the ideal balance of patients/consumers and healthcare professionals on guideline panels, composition of non-professional stakeholders (e.g. patients, caregivers, advocates, consumers), and potential practical measures for improved involvement.

Given this gap, we performed a pilot study with focus groups of individuals not involved in guideline development in preparation for a larger study on patient engagement in guideline development. The aim of the pilot study was to explore guideline-naïve individuals’ preferences for engagement in guideline development if invited, including topics relating to panel composition, barriers to successful engagement, and proposed strategies to enhance meaningful participation.

A total of 15 individuals participated over three focus groups and one key informant interview. The majority of participants were female and/or African-American with a diverse representation of ages (Table 1). Participants largely viewed themselves participating in guideline panels as patient representatives with disease-specific experience rather than as consumers, so the term “patients” is used throughout the results except where consumer involvement was specifically discussed. The focus groups at the University of Maryland each used less time than allotted; the University of Florida focus group was able to address each of the planned questions within the full allotted time. Results from the focus groups are presented here in themes emerging from the analysis.

This study suggests that ideal guideline panels are small and engage multiple patient/consumer stakeholders (Table 3). Diversity is important with regards to engaging individuals with different disease perspectives, ethnicities, and roles (patients, consumers, advocates, and healthcare professionals of various backgrounds). Given that some participants described discomfort with certain situations (e.g. mixing patients and caregivers on a panel), potential participants should know anticipated panel composition in advance. In general, the focus groups preferred anonymity. To facilitate patient/consumer engagement, guideline developers should ensure that meetings are conducted using language that all can understand and with sufficient time allotted for sharing opinions and discussion. Providing writing materials is a practical way to help panel members note thoughts to share. When recruiting patients and consumers, guideline developers should establish the purpose of their engagement and provide preparatory materials and a point of contact. Facilitators should be sensitive, skilled moderators, and able to engage all panel members, possibly with pre-established plans for soliciting views, particularly if panel members are hesitant to volunteer opinions. Multiple in-person meetings may build comfort but have logistical challenges.

These results are consistent with prior publications. Various groups recommend multiple patient/consumer stakeholders [4, 8, 9, 19]; isolation is a barrier to successful patient engagement [8]. Diversity in panel composition is also recommended by the Institute of Medicine [5], G-I-N [3], and AGREE2 [20]. Medical terminology is a well-established barrier to successful patient engagement [7–9] and was also highlighted in our study. Perceived barriers of physician resistance to patient participation and personal lack of content knowledge are consistent with known barriers [8]. Establishing the purpose of patient participation in guideline panels is described as a facilitator of successful engagement in other reports [4, 8]; providing pre-reading materials can address pre-meeting apprehension [4] and can serve as one method of advanced training to facilitate engagement [4, 8]. Use of a “welcome pack” is described as a facilitator to successful patient engagement and is a method of providing both training and support [8]. The importance of engaging a skilled chair/moderator is a point made by our participants and others [1, 4, 8].

New implications include an emphasis on the logistical and practical measures that guideline developers can take to enhance patient engagement (Table 3), something not captured in a recent patient engagement framework. Additionally, to our knowledge, this study is the first to explore the preferences regarding pre-meeting relationships of panel members and issues regarding race and diversity. While medical terminology is a well-established barrier to successful patient engagement, the barrier of difficulty understanding spoken language (i.e., accents) was also identified by multiple participants. Lack of sufficient time for meetings/collaboration is a newly identified (though hardly surprising) barrier that developers can address. Finally, study participants made the point that guideline developers and panels should assess panel results and products to ensure that the patient voice was adequately captured and avoid tokenistic engagement.

While not directly suggested by focus group participants, a strategy implied by the findings is the importance of educating potential participants prior to their agreement to join a guideline development group (Table 3). This makes sense given limited public knowledge regarding guidelines and guideline development [15, 21]. In the guideline-naïve participants in this study, there were misconceptions about guidelines and the guideline process such that some suggestions were impractical (such as patients greatly outnumbering professional participants on a panel). This suggests that potential participants may not understand the process, goals, and scope of involvement and this should be clarified both before and after consent to participate. Additionally, given that some participants described discomfort with certain situations (e.g. mixing patients and caregivers on a panel), potential participants should know anticipated panel composition in advance. This advanced education is in addition to training individuals who have already agreed to participate, a well-described facilitator to engagement [1, 3, 4, 8, 13].

Limitations of the present study include the fact that participants had no experience with guideline development and minimal knowledge of guidelines prior to participating in the focus groups. Thus, they were making judgments and recommendations based on hypothetical engagement. However, developers recruiting patients for guideline panels are likely to largely encounter patients or members of the public with no knowledge of guidelines, making these findings particularly helpful for developers preparing to promote and support the involvement of guideline-naïve patients/public in the process. It is notable that many comments and suggestions by our guideline-naïve participants reflect barriers and facilitators described in the literature by those with guideline expertise. This underscores the power of obtaining the patient view–these participants were able to identify in advance barriers and facilitators to engagement that developers are learning by reflecting on initial experiences. Naïve statements and an identified lack of understanding regarding some aspects of guideline development also informed some key suggestions (Table 3), such as training as part of recruitment rather than solely after an agreement to participate.

The lack of understanding reflected in some responses could also result from the description of guideline development at the start of the focus groups (S1 File), which was intentionally kept simple to enhance understanding by participants with varying health literacy. Guideline methodologists and participants drafted and revised outline content and edits to enhance patient and consumer understanding were implement by PATIENTS Program staff, but the introduction did not capture all nuances of guideline development nor the breadth of different developers’ approaches. Given the overlap between our results and those in the literature, participants appeared to understand many aspects of guideline development based on the introductory material. Some impractical suggestions, such as patients and consumers greatly outnumbering professionals on guideline panels, persisted even after clarification of typical guideline group composition (e.g. professionals representing different areas of expertise, such as content vs methodology expertise). Thus, these unexpected views are important for guideline developers to consider, both in terms of adequate engagement and recruitment training.

We did not solicit feedback on published barriers and facilitators to patient engagement, preferring instead to allow participants to generate their own thoughts, particularly as we were investigating the views of guideline-naïve potential participants (in contrast to currently published literature). This means that some well-accepted facilitators to engagement, such as training, were not explored in our focus groups. These focus groups reflect a pilot study preparing for a larger project investigating the impact of engaging patients, caregivers, and advocates on guideline question development and as such, only two patient organizations were targeted. This means that saturation of themes may not have been achieved. Participants agreed to one-time group participation and not future contact, so feedback on transcripts and themes was not obtained from participants subsequent to the focus groups. Finally, while attempts were made to obtain diversity in age, gender, and ethnicity, the number of participants and focus group conduct in two locations in the United States means that these findings are potentially not transferable to patients in other locations.

Future research should further explore the implications of panel composition including issues of race and racial diversity, the impact of engaging non-physician professionals, and the optimal balance of lay and professional participants. Research is also warranted into logistical issues, such as the impact of meeting type (in-person, telephone, webinar, all-electronic) and frequency. Understanding optimal strategies for engagement will require further insight into these practical considerations in addition to the topics of strategic recruitment, relationship-building, co-learning, and reassessment. It is also important to understand if certain factors have more impact on successful engagement such that they should be prioritized by guideline developers. Finally, future research could directly compare views of participants with and without guideline experience or investigate views of guideline-naïve individuals before and after guideline development group participation to further understand how views differ between those individuals with and without guideline experience.

This study adds an additional voice to the research regarding patient engagement in guideline development panels: that of individuals not yet recruited. Emerging recommendations for successful engagement overlap with existing literature but also provide new strategies, including highlighting the importance of practical and logistical issues. Guideline developers should consider small, diverse panels with multiple patient/consumer stakeholders having no prior relationships. Providing goals for participation, pre-meeting reading/training and a point of contact are keys to successful early engagement; skilled facilitators, an emphasis on understandable speech and language, and established mechanisms for soliciting patient opinions enhance patient engagement at meetings. Most of these strategies are inexpensive, an important consideration for many guideline developers.