Patient and Public Involvement in Inflammatory Bowel Disease Research—A Scoping Review

Abstract

Background

Interest in patient and public involvement in research has grown. Medical, health, and social care research has demonstrated several benefits of patient and public engagement, such as empowering user input and reducing attrition rates in clinical trials. To date, no study has reviewed patient engagement in inflammatory bowel disease (IBD). We aimed to describe the benefits, challenges, and best practices of patient engagement in IBD research.

Methods

We performed a systematic search on MEDLINE, EMBASE, and Cochrane for all clinical IBD research studies in which patients were involved in the research process (1946- 2023). Patient input was considered in: (1) study design, (2) study execution, (3) research dissemination, and/or (4) other domains not specified here. Two authors independently screened and extracted data on type of engaged person(s), format of engagement, author-reported benefits, recommendations, and challenges. For each study, we reported the level of patient engagement and study adherence to standardized reporting guidelines.

Results

After screening 9,355 articles, we included 51 for final analysis. IBD patients were most frequently engaged in study design. Patient engagement in IBD research improved recruitment rates and promoted the creation of user-friendly quality-of-life tools. Selection bias and recruitment difficulties were common challenges in the application of patient engagement. Authors recommended continuous patient involvement to address emerging priorities and cognitive interviewing to improve questionnaire clarity.

Conclusions

Patient engagement represents an important step in promoting patient-centred care. According to study authors, implementing cognitive interviewing techniques, continuous patient involvement, and standardized reporting guidelines may improve future iterations of engagement in IBD.

Introduction

Interest in patient and public involvement (PPIn) in research has grown.¹ An early example of patient involvement occurred in the 1980s in the UK, wherein patient group representatives conducted a trial on chorionic villous sampling in pregnancy.² Stakeholder engagement has also been adopted in social and healthcare research in the UK, where the INVOLVE project supports active public involvement in the National Health Service (NHS).³ Since then, there has been a growing emphasis on engaging patients in research directly related to their care.⁴

Research in health and social care has demonstrated several benefits of PPIn.^5,6 These include empowering user input and providing researchers with a greater understanding of community perspectives.⁶ In medicine, PPIn has been demonstrated to positively impact patient enrolment in clinical trials.¹ On ethical grounds, engaging patients in research relevant to their care may democratize the research process.⁷ To date, no study has comprehensively reviewed patient engagement in inflammatory bowel disease (IBD) research. IBD is a chronic disease with a significant burden on patients’ personal, social, and occupational functioning.⁸ IBD patients frequently play an active role in their disease management.⁸ As such, IBD patients may be able to offer their knowledge to research studies as experience-based experts.

In this scoping review, we analyzed studies that actively involved IBD patients (and other relevant stakeholders) in research. Using a meta-narrative approach,⁹ we aimed to provide a synthesis on implementing patient engagement in IBD and to address the following questions: How and during what stage of research are IBD patients engaged? What are author-reported recommendations for best practices of PPIn in IBD? What are the benefits of PPIn in IBD? What are the challenges (and suggested solutions) of PPIn in IBD?

Methods

We comprehensively reviewed IBD research studies in which patients were active participants in the research process. Reporting of our findings followed guidelines for the reporting of scoping reviews (PRISMA extension for Scoping Reviews).¹⁰ A completed checklist of the reporting guidelines can be found in Text, Supplementary Data Content 1.

Search strategy

We ran a systematic search in EMBASE, Medline, and the Cochrane Central Register of Controlled Trials for clinical IBD research studies in which patients were actively involved in research. The following study types were considered for inclusion: clinical practice guidelines, randomized controlled trials, cohort studies, cross-sectional studies, systematic reviews, meta-analyses, survey studies, cost-effectiveness analyses, and qualitative studies. We excluded opinion pieces, editorials, abstract-only submissions, research letters, case reports/series, and protocol-only entries. We only included articles published in English. The search was not limited to specific countries. The search was designed by a librarian. The search strategy was also peer-reviewed and assessed against the Peer Review of Electronic Search Strategy (PRESS) checklist.¹¹ The searches were run from inception to August 21, 2023. The full search strategies can be found in Text, Supplementary Data Content 2.

Study selection

Two authors independently reviewed the abstracts (K.E., E.N.) and full texts (K.E., A.L.) for eligible studies using the screening platform Covidence. Discrepancies were resolved by consensus. Patient input was considered in: study design (prioritization of research topics, outcome selection, study tool development, and funding acquisition); study execution (study recruitment, data collection, and analysis); dissemination of research (manuscript development); and/or other domains not specified here. Engaged person(s) included IBD patient(s), and/or other stakeholders (patient representatives, caregivers, and community members).

Data extraction

Two authors extracted the following data from full texts: article type, study objectives, research setting, enrolment method(s), sampling method(s), engaged person(s), and method(s) of PPIn, which included focus groups, semi-structured interviews, surveys, and/or multidisciplinary panels. Where possible, we sorted instances of PPIn into one or more of the following categories: study design, study execution, and/or dissemination of results.

For each instance of PPIn, we obtained a narrative description of patient contributions from the study article. We searched each full text for author and patient-reported best practices, benefits, challenges (and suggested solutions) of PPIn. We reported level of patient engagement according to the Patient-Oriented Research Level of Engagement Tool (PORLET) and adherence to standardized reporting guidelines according to the Guidance for Reporting Involvement of Patients and the Public, Version 2 (GRIPP2) for each study.^12,13

Analysis

Quantitative data on article type, engaged person(s), method(s) of engagement, and stage of patient engagement in the research process was summarized using descriptive statistics. The qualitative data were analyzed using a meta-narrative approach, adopted from Domecq et al.^7,9 Following a literature search with clear selection criteria, the approach adheres to a framework defining four key questions (methods, best practices, benefits, and challenges of PPIn). As per the framework, we reviewed narrative data from each article and synthesized key themes, using specific studies to highlight themes and map available evidence. Specifically, we reviewed the Discussion section of each article for methods, benefits, challenges, and best practices of PPIn as identified from prior literature,^7,14 and extracted any additional themes reported by the authors.

Results

Our search identified 9,355 articles. Following title and abstract screening and full-text review, we included 51 articles (Figure 1). Most articles were qualitative studies (n = 41, 80.4%). Our sample also included four survey studies (7.8%), two randomized controlled trials (RCTs) (3.9%), three retrospective cohort studies (5.9%), and one cross-sectional study (2.0%). Thirty-three percent (n = 16) of patient engagement studies were based in the UK, and the majority (n = 48, 94.1%) were academic-affiliated.

Figure 1.

Study flow chart.

Most studies involved IBD patients directly (n = 50, 98%). Remaining engaged parties included patient representatives (n = 14, 27.4%), caregivers (n = 10, 19.6%), and community members (n = 1, 2.0%). Patients were engaged via semi-structured interviews (n = 24, 47.1%), multi-disciplinary panels (n = 27, 52.9%), focus groups (n = 22, 43.1%), and surveys (n = 13, 25.5%). Patients were engaged in study design in most studies (n = 50, 98%), study execution in 22 studies (43.1%), and dissemination of results in nine studies (17.6%). A summary of article characteristics can be found in Table 1.^15–65

Table 1.

Study characteristics and level of engagement according to Patient-Oriented Research Level of Engagement Tool (PORLET) criteria (n = 51).

Article	Article Year	Article objective	Article type	Study institution (country)	Study institution (type)	Engaged person(s)	Method of engagement	Timing of engagement (study Phase)	PORLET Score (/25)
Adegbola et al.15	2021	Patient-reported outcome measures	Qualitative study	United Kingdom	Academic hospital	Patient, patient representative	Semi-structured interviews	Study design, study execution	14
Adegbola et al.16	2020	Thematic analysis	Qualitative study	United Kingdom	Academic hospital	Patient, patient representative	Multi-disciplinary panels	Study design	12
Adler et al.17	2016	Thematic analysis	Qualitative study	United States	Academic hospital	Patient, caregiver	Surveys, tool development	Study design	11
Alnaqbi et al.18	2013	IBD instrument development	Qualitative study	Canada	Academic hospital	Patient	Surveys, semi-structured interviews	Study design	7
Alrubaiy et al.19	2015	IBD instrument development	Qualitative Study	United Kingdom	Academic hospital	Patient	Surveys, semi-structured interviews, focus groups	Study design	8
Bitton et al.20	2020	IBD instrument development	Qualitative Study	Canada	Academic hospital	Patient, patient representatives	Focus groups, multi-disciplinary panels	Study design	8
Bitton et al.21	2019	Quality indicator assessment	Qualitative study	Canada	Academic hospital	Patient, patient representative	Focus groups, multi-disciplinary panels	Study design	10
Bodger et al.22	2014	IBD instrument development, IBD instrument validation	Qualitative study	United Kingdom	Academic hospital	Patient	Focus groups, semi-structured interviews	Study design	11
Carter et al.23	2020	Thematic analysis	Qualitative study	United Kingdom	Academic hospital	Patient, caregiver	Surveys, semi-structured interviews	Study design, study execution, dissemination of results	14
Casellas et al.24	2013	IBD instrument Development, IBD instrument Validation	Qualitative study	Spain	Academic hospital	Patient	Semi-structured interviews	Study design	7
Cheifetz et al.25	2012	IBD research priorities	Qualitative study	United States	Academic hospital	Patient	Focus groups	Study design	10
de Jong et al.26	2017	IBD instrument development	Qualitative study	Netherlands	Academic hospital	Patient	Multi-disciplinary panels	Study design	17
Denters et al.27	2012	IBD instrument development	Qualitative study	Netherlands	Academic hospital	Patient	Focus groups	Study design	9
Dibley et al.28	2018	IBD instrument development, IBD instrument validation	Qualitative study	United Kingdom	Academic hospital	Patient	Semi-structured interviews	Study design, study execution	14
Dibley et al.29	2020	IBD instrument development, qualitative analysis	Qualitative study	United Kingdom	Academic hospital	Patient	Multi-disciplinary panels	Study design, study execution, dissemination of results	14
Fofaria et al.30	2019	Quality improvement project	Qualitative study	United Kingdom	Academic hospital	Patient, patient representative	Multi-disciplinary panels	Study design, study execution	20
Grant et al.31	2019	IBD research priorities	Qualitative study	Canada	Academic hospital	Patient, caregiver	Focus groups, multi-disciplinary panels	Study design, study execution	23
Guida et al.32	2021	Thematic analysis	Qualitative study	Italy	Academic hospital	Patient	Focus groups	Study design	7
Haaland et al.33	2014	IBD instrument development, IBD instrument validation	Qualitative study	Canada	Academic hospital	Patient, caregiver	Focus groups, semi-structured interviews	Study design	7
Hart et al.34	2017	IBD research priorities	Qualitative study	United Kingdom	Academic hospital	Patient, patient representative	Multi-disciplinary panels	Study design, study execution	17
Hubbard et al.35	2019	IBD instrument development	Qualitative study	United Kingdom	Academic hospital	Patient	Multi-disciplinary panels	Study design, study execution, dissemination of results	16
Hughes et al.36	2016	IBD instrument development, IBD instrument validation	Survey with high response rate	United Kingdom	Academic hospital	Patient	Semi-structured interviews	Study design	13
Kapasi et al.37	2020	IBD instrument development	Qualitative study	United Kingdom	Academic hospital	Patient, patient representative, caregiver	Focus groups, multi-disciplinary panels	Study design	16
Katarina et al.38	2018	IBD instrument development	Qualitative study	Sweden	Academic hospital	Patient	Surveys, semi-structured interviews, focus groups	Study design	10
Kennedy et al.39	2003	IBD instrument development	Intervention studies—RCT	United Kingdom	Academic hospital	Patient, patient representative	Focus groups, multi-disciplinary panels	Study design	13
Kennedy et al.40	2002	IBD instrument development	Qualitative study	United Kingdom	Academic hospital	Patient	Semi-structured interviews	Study design	11
Kennedy et al.41	2019	IBD instrument development	Qualitative study	United States	Academic hospital	Patient, patient representative	Multi-disciplinary panels	Study design, study Execution, dissemination of results	22
Khalil et al.42	2020	IBD instrument development	Qualitative study	United States	Academic hospital	Patient	Semi-structured interviews, focus groups	Study design	13
Kim et al.43	2021	IBD unstrument Development	Qualitative study	Australia	Academic hospital	Patient	Surveys, semi-structured interviews, focus groups, multi-disciplinary panels	Study design	10
Kim et al.44	2018	IBD Instrument Development	Qualitative study	Australia	Academic hospital	Patient	Semi-structured interviews, focus groups, multi-disciplinary panels	Study design	20
Lee et al.45	2021	IBD Instrument Development	Qualitative study	United Kingdom	Academic hospital	Patient, patient representative	Surveys, semi-structured interviews, multi-disciplinary panels	Study design	8
Louis et al.46	2020	IBD Research Priorities	Qualitative study	Belgium	Academic hospital	Patient, patient representative	Focus groups, multi-disciplinary panels	Study design, study execution	17
Wickman et al.47	2018	IBD Instrument Development	Qualitative study	Sweden	Academic hospital	Patient	Surveys, semi-structured interviews	Study design, study execution	8
Macdonald et al.48	2018	Thematic Analysis	Observational studies—Cross-sectional study	Canada	Academic hospital	Patient	Surveys, multi-disciplinary panels	Study design, study execution	17
Marín-Jiménez et al.49	2017	IBD Instrument Development	Survey with high response rate	Spain	Academic hospital	Patient, patient representative	Surveys, focus groups, multi-disciplinary panels	Study design, study execution	17
McDermott et al.50	2018	Patient Education in IBD	Qualitative study	Ireland	Academic hospital	Patient	Surveys, focus groups	Study design, Study execution	12
Oliver et al.51	2021	IBD Instrument Development	Qualitative study	United States	Academic hospital	Patient	Multi-disciplinary panels	Study design	9
Rohatinsky et al.52	2020	Thematic Analysis	Qualitative study	Canada	Community hospital	Patient, caregiver	Semi-structured interviews, multi-disciplinary panels	Study design, study execution, dissemination of results	25
Ruan et al.53	2017	IBD instrument development, IBD instrument validation	Qualitative study	China	Academic hospital	Patient	Surveys, semi-structured interviews	Study design	8
Vent-Schmidt et al.54	2020	Thematic analysis	Qualitative study	Canada	Academic hospital	Patient	Semi-structured interviews, focus groups	Study design	8
Vergara et al.55	2002	IBD instrument development, IBD instrument validation	Survey with high response rate	Spain	Academic hospital	Caregiver, community member	Semi-structured interviews	Study design	7
Williams et al.56	2021	IBD instrument development	Qualitative study	Australia	Academic hospital	Patient, patient representative	Surveys, semi-structured interviews, focus groups	Study design	18
Sahnan et al.57	2019	Patient-reported outcome measures	Qualitative study	United Kingdom	Academic hospital	Patient, patient representative	Semi-structured interviews, multi-disciplinary panels	Study design, study execution	13
Heisler et al.58	2023	Thematic analysis	Qualitative study	Canada	Community hospital	Patient	Multi-disciplinary panels	Study design, study execution	16
Santos et al.59	2022	Health systems research	Observational studies—cohort study	Canada	Community hospital	Patient, caregiver	Multi-disciplinary panels	Study design, study execution, Dissemination of results	20
Cavallaro et al.60	2021	Patient-reported outcome measures	Qualitative study	New Zealand	Academic hospital	Patient	Focus groups, multi-disciplinary panels	Study execution	7
Rines et al.61	2021	Thematic analysis	Qualitative study	Canada	Academic hospital	Patient	Multi-disciplinary panels	Study design, study execution, dissemination of results	25
Long et al.62	2023	IBD instrument development	Intervention studies—RCT	United States	Academic hospital	Patient	Semi-structured interviews, focus groups	Study design	8
Gorbenko et al.63	2021	IBD instrument development	Qualitative study	United States	Academic hospital	Patient	Focus groups	Study design	8
Peña-Sánchez et al.64	2022	Prevalence study	Observational studies—Cohort study	Canada	Academic hospital	Patient, caregiver	Multi-disciplinary panels	Study design, dissemination of results	20
Peña-Sánchez et al.65	2022	Health systems research	Observational studies—Cohort study	Canada	Academic hospital	Patient, Caregiver	Semi-structured interviews, multi-disciplinary panels	Study design, study execution, dissemination of results	25

How are IBD patients engaged in the research process?

The most frequent form of PPIn was patient consultation in the development of IBD study tools, including questionnaires, guidebooks, and a video animation (n = 44, 86.3%). A qualitative study exploring disclosure of IBD among children utilized a core e-Advisory group of IBD patients to provide insight into young people’s experiences with disclosure,²³ which informed the development of a two-minute animation.²³

IBD patients and their surrogates also acted as active participants in study recruitment in four studies (7.8%).^15,35,52,65 In one study, patient champions within the project team devised a new method of identifying and transferring IBD patients to specialist nurse-led telephone clinics, which helped boost transfer rates.³⁰

Other modalities of PPIn included assisting with data analysis (n = 8, 15.7%), wherein one study provided patients with training in qualitative analysis methods from an experienced qualitative researcher.²⁹ Six studies (11.8%) included patients and/or other stakeholders (e.g., patient advisors) as co-authors on the published work. Of note, one study on the development of an IBD transfer toolkit was spearheaded by an IBD patient, who acted as the lead study author in collaboration with a paediatric gastroenterologist.⁴¹ This was achieved by involving the patient partner from the outset of the project in study design. One retrospective study directly cited engaged patients’ reflections on study results in their discussion, including their thoughts and assessment of the increasing prevalence of IBD among First Nations.⁶⁴

Adherence to reporting guidelines for PPIn was evaluated via the GRIPP2 checklist (Table 2). All studies in our cohort (n = 51) reported the methods used for PPIn. Twenty-five reported the aim of PPIn, 35 reported results of PPIn, 32 reported positive and/or negative effects of PPIn, and 19 commented critically on PPIn in the study (reflecting on positive experiences and/or areas of improvement).

Table 2.

PPIn according to guidance for reporting involvement of patients and the public, version 2 (GRIPP2) reporting checklist (short form) (n = 49).

Article	1. Aim	2. Methods	3. Results	4. Discussion/conclusions	5. Reflections/critical perspective
Adegbola et al.15		X	X	X
Adegbola et al.16	X	X	X	X
Adler et al.17		X
Alnaqbi et al.18		X	X
Alrubaiy et al19		X	X
Bitton et al.20	X	X	X
Bitton et al.21		X	X	X	X
Bodger et al.22	X	X	X	X	X
Carter et al.23		X	X
Casellas et al.24		X	X	X	X
Cheifetz et al.25		X			X
de Jong et al.26		X
Denters et al.27	X	X	X	X	X
Dibley et al.28	X	X	X	X
Dibley et al.29		X	X	X
Fofaria et al.30		X			X
Grant et al.31	X	X	X	X	X
Guida et al.32		X
Haaland et al.33		X
Hart et al.34	X	X	X	X	X
Hubbard et al.35	X	X
Hughes et al.36		X	X	X
Kapasi et al.37	X	X	X	X
Katarina et al.38	X	X	X	X	X
Kennedy et al.39		X	X	X
Kennedy et al40	X	X		X
Kennedy et al.41	X	X	X	X
Khalil et al.42	X	X	X	X	X
Kim et al.43		X	X	X
Kim et al.44	X	X	X	X	X
Lee et al.45		X
Louis et al.46		X	X	X	X
Wickman et al.47		X	X	X	X
Macdonald et al.48	X	X		X	X
Marín-Jiménez et al.49		X	X	X
McDermott et al.50	X	X	X	X
Oliver et al.51		X	X	X
Rohatinsky et al.52	X	X	X	X
Ruan et al.53		X
Vent-Schmidt et al.54	X	X
Vergara et al.55		X	X	X
Williams et al.56	X	X			X
Sahnan et al.57		X	X	X	X
Heisler et al.58	X	X
Santos et al.59		X	X	X
Cavallaro et al.60	X	X	X	X
Rines et al.61	X	X	X	X	X
Long et al.62	X	X
Gorbenko et al.63		X	X	X	X
Peña-Sánchez et al.64	X	X	X		X
Peña-Sánchez et al.65	X	X

Level of engagement

Level of patient engagement was reported for each study according to the PORLET criteria, which gauges the degree of PPIn according to five criteria (Table 1). Score breakdown by individual criteria is available in Text, Supplementary Data Content 3. The mean total PORLET score was 13.3 (SD 5.4). There was a wide range of PORLET scores reported,^7–25 pointing to heterogeneity in the level of engagement.

A pair of studies linked to a patient-oriented research initiative achieved a high level of engagement.^52,65 One study evaluated perspectives of healthcare use among rural Canadians with IBD.⁵² The study engaged patient and family advisors as equal members in decision-making. This was achieved by engaging patients in all phases of the research process. Patient partners contributed to the study design, co-created recruitment materials, reviewed findings, and co-developed the manuscript.^52,65 A separate study with high patient engagement compared healthcare utilization between First Nations patients and the general IBD population. This retrospective cohort study involved indigenous patients in study outcome selection, data analysis, and the knowledge-sharing phases of research (wherein 1 advocate co-presented the published work at multiple conferences).⁵⁹ This was mediated by sending the engaged parties periodic reports and requesting regular feedback on results.⁵⁹

What are author-reported recommendations for best practices of PPIn in IBD?

Three IBD studies in our sample (5.9%) cited the importance of recruiting patients broadly for PPIn.^15,24,36 In one study, authors noted that recruiting patients from a wide variety of centres and different regions of Spain helped ensure the generalizability of their findings.²⁹ Two studies (3.9%) encouraged cognitive interviewing with patients as a tool in questionnaire development, with a validation study noting it improved question clarity, ease of understanding, and increased item validity.^38,47 Last, a study engaging IBD patients in online survey development recommended continuous patient involvement to address patients’ emerging concerns during clinical testing.⁵⁴

What are the benefits of PPIn in IBD?

The most reported benefit of PPIn, cited across 21 studies (41.2%), was that it helped promote patient-centred care and/or ensured that the patient voice was represented. In one study soliciting research uncertainties in the management of pediatric IBD, authors highlighted the role of patient engagement in establishing a research agenda that was mutually impactful to both those treating and those experiencing the disease.³¹

A second theme centred on the role of PPIn in strengthening the relevance and/or credibility of study items. Authors of a quality-of-life questionnaire for household members of IBD stated that involving IBD stakeholders in the development of their tool helped focus items on issues most relevant to responders.⁵⁵ Lastly, a tertiary benefit reported by one study was that patient engagement promoted the creation of a user-friendly and understandable quality-of-life tool in IBD research.¹⁹

What are the challenges (and suggested solutions) of PPIn in IBD?

Two studies (3.9%) cited feasibility as a challenge to incorporating PPIn in IBD research, specifically as it relates to recruiting and engaging participants as part of study design.^25,56 In a study testing a decision aid encompassing reproductive decisions in IBD, authors made note of intrinsic difficulties with patient recruitment for PPIn, related to the time required to conduct patient focus groups and guide the development of the decision aid. This was cited as a challenge in recruiting patient participants, especially young patients who may have professional commitments and/or time constraints related to family.⁵⁶ Another challenge identified across 4 studies (7.8%) was that engaged patients may not be representative of the entire IBD population. One study highlighted a lack of diversity in education and literacy among focus group participants.⁴² The authors of a quality-of-care questionnaire developed by Swedish IBD patients stated that the use of the questionnaire may be limited to a Swedish care context.³⁸ These authors recommended cultural adaptation of the tool if it were to be used in a different context.

Discussion

In our analysis of 51 original articles involving patients and/or other stakeholders in IBD research, we found that IBD patients were most frequently engaged in the design phase of the research process. Across three studies, they were involved in setting IBD research priorities. The influence of PPIn on the research agenda has previously been reported in a review of INVOLVE, a publicly funded NHS patient involvement project.¹⁴ Several studies in the review demonstrated that PPIn led to clearer research priorities, a wider set of topics being considered, and a shift in research focus to be more in line with public concerns.^14,66 More recently, a summative report on the impact of IBD in Canada incorporated patients’ and caregiver partners’ perspectives on several research articles.⁶⁷ The goals of this engagement were to define key advocacy priorities for an IBD patient organization and to highlight content within the report that was most relevant to IBD patients.⁶⁷

In one study by McCormick et al., the involvement of women with breast cancer in environmental causes research shifted the focus of the program from a biochemical model to a political one that better reflected the women’s interests.⁶⁸

PPIn was also cited to enhance the credibility of IBD study tools. This effect has been demonstrated in community and social care research, where evidence suggests public engagement gives both the researchers and the research project greater credibility.^7,69 For instance, in a research study involving Alaska Natives, community members outperformed researchers in recruiting participants. This was attributed to community members giving legitimacy to claims the project was being conducted in partnership with the community.⁷⁰ Several factors may explain the observed role of PPIn in improving research enrolment. First, as seen in three studies in our cohort, patients may act as active participants in study recruitment, directly supporting the efforts of study authors. Second, patients may indirectly support study recruitment via contributions to research design. Authors of a pediatric RCT on osteopathy attributed the trial’s high enrolment rate to the stakeholders’ contributions to group design.⁷¹ A RCT on environmental control education in asthma similarly acknowledged the role of a community advisory board in promoting successful recruitment strategies, along with creating study reports that were understandable to the community.⁷²

With these benefits in mind, the costs of PPIn, along with recruitment difficulties, may limit the feasibility of incorporating PPIn in IBD research. A literature review by Shah et al. cited time constraints as a key impediment to involving users in the development and evaluation of medical device technologies.^73,74 PPIn likely represents a significant time commitment for patients and researchers, which may hinder its widespread application. The profile of engaged patients may also differ substantially from that of the general IBD population. Viswanathan et al. (2004) cited a recruitment bias toward highly motivated people in research involvement. In the IBD cohort, such a bias might hamper the applicability of certain quality-of-life interventions, which often require significant investment on the part of the patient.⁵

Study heterogeneity in our sample limits our synthesis of patient engagement in IBD. Best practices of PPIn are likely to vary with the context of engagement. Nevertheless, we outline the following recommendations for implementing participatory research frameworks in IBD. (1) We recommend recruiting engaged parties broadly to ensure findings are generalizable to the IBD population. (2) With time commitment being a key limitation in recruitment, we encourage researchers to elicit regular patient feedback to modify the involvement process in accordance with patient needs. (3) When engaging patients in questionnaire development, we propose employing cognitive interviewing techniques to improve individual item clarity. (4) We recommend engaging patients continuously throughout the research process to address emerging concerns. This is consistent with prior research from Lindenmeyer et al. and Rhodes et al., who found long-term involvement aided user-researcher cohesion and facilitated a deeper knowledge of and contribution to the research process.^69,73 (5) We recommend authors adhere to reporting guidelines for PPIn such as PORLET and GRIPP2 to better define the scope of PPIn and standardize its methodology.

In addition to the above recommendations, the Canadian Institutes of Health Research has designed a framework highlighting guiding principles, and core areas of patient engagement for adoption by clinical research partners.⁷⁵ The Strategy for Patient-Oriented Research highlights guiding principles for patient engagement such as integrating PPIn into researcher training, implementing tangible incentives, creating a diverse pool of patient participants, and securing financial compensation to support PPIn.⁷⁵

We note several study limitations. Our search likely did not capture all instances of PPIn in IBD, as study authors may not strictly adhere to standardized reporting guidelines defining PPIn such as GRIPP2. In addition, reporting biases may have been present in our sample. The benefits, challenges, and best practices of PPIn in IBD were reported directly by authors and, accordingly, may be subject to several biases, including pre-existing conceptions of the role of PPIn in research.⁷ In the absence of a robust measurement of impact, our interpretation of these findings is limited.⁷⁶ Similar limitations were noted in a 2009 review exploring the impact of public involvement in the NHS, citing a lack of quantitative evidence of benefit, which is largely based on the views of researchers and members of the public.¹⁴ We were also unable to identify any benefits or challenges reported by engaged patients, who offer a valuable perspective and may represent a hypothesis generating future research ideas. We excluded certain study types (e.g., opinion pieces and editorials), which may feature PPIn. Lastly, the pool of engaged person(s) in our cohort may have experienced selection bias. Due to time availability among other factors, certain IBD patients may be more likely to be recruited.³⁴

Considering these limitations, we highlight the implications of our findings. PPIn represents an important step in advancing patient-centred care. Moreover, authors highlighted several recommendations that may be incorporated in future iterations of PPIn in IBD, such as cognitive interviewing and continuous patient involvement.

Conclusions

PPIn in IBD improved recruitment rates, promoted the creation of user-friendly quality-of-life tools, and ensured patients’ concerns were reflected in the research process. Authors recommended continuous involvement of patients throughout the research process to address their emerging priorities. PPIn in IBD may be limited by recruitment difficulties and selection bias. Future research in this area should focus on assessing the impact of patient engagement on the efficacy of quality-of-life metrics in IBD.

Supplementary data

Supplementary data are available at Journal of the Canadian Association of Gastroenterology online.

Author Contributions

Study conception and design: K.E., S.S., R.K., T.K., M.H., M.A.S., N.K., S.C.G. Data acquisition: K.E., A.L., M.H., E.N. Analysis and interpretation of data: K.E., A.L., S.C.G.Drafting of the manuscript: K.E., M.A.S., N.K., R.K., S.C.G.Study supervision: S.C.G.

Funding

The authors received no specific funding for this study.

Conflict of interest

Rishad Khan has received research grants from AbbVie and Ferring Pharmaceuticals and research funding from Pendopharm. Samir C. Grover has received research grants and personal fees from AbbVie and Ferring Pharmaceuticals, personal fees from Takeda, education grants from Janssen, and has equity in Volo Healthcare. All other authors have no relevant disclosures.

Data availability

The data underlying this article are available in the article and in its supplement and where used is cited under References. Additional data may be shared upon request.

Conference presentations

The present study was presented as a poster in Canadian Digestive Diseases Week 2022 and in Digestive Diseases Week 2023.