How and why should we engage parents as co‐researchers in health research? A scoping review of current practices

Abstract Background The importance of engaging parents in health research as co‐researchers is gaining growing recognition. While a number of benefits of involving parents as co‐researchers have been proposed, guidelines on exactly how effective engagement can be achieved are lacking. The objectives of this scoping review were to (i) synthesize current evidence on engaging parents as co‐researchers in health research; (ii) identify the potential benefits and challenges of engaging parent co‐researchers; and (iii) identify gaps in the literature. Methods A scoping literature review was conducted using established methodology. Four research databases and one large grey literature database were searched, in addition to hand‐searching relevant journals. Articles meeting specific inclusion criteria were retrieved and data extracted. Common characteristics were identified and summarized. Results Ten articles were included in the review, assessed as having low‐to‐moderate quality. Parent co‐researchers were engaged in the planning, design, data collection, analysis and dissemination aspects of research. Structural enablers included reimbursement and childcare. Benefits of engaging parent co‐researchers included enhancing the relevance of research to the target population, maximizing research participation and parent empowerment. Challenges included resource usage, wide‐ranging experiences, lack of role clarity and power differences between parent co‐researchers and researchers. Evaluation of parent co‐researcher engagement was heterogeneous and lacked rigour. Conclusions A robust evidence base is currently lacking in how to effectively engage parent co‐researchers. However, the review offers some insights into specific components that may form the basis of future research to inform the development of best practice guidelines.

that family members are usually the constant in a child's life and as such represent considerable expertise in the child's health and care. [2][3][4][5] The concept of engaging patients and their families in health research greatly aligns with the principals and core beliefs of CFCC and has also recently gained ground and attracted the attention of researchers and policymakers. [6][7][8][9] Government funding agencies have created specific mechanisms through which to promote patient engagement in research, such as the Strategy for Patient-Oriented Research initiative in Canada and the Food Drug Administration Patient Engagement Advisory Committee in the United States. However, while systematic reviews have focused upon involving patients and their families in research generally, 8,10 none have looked at parents specifically. While it is vital to engage well children in research, parents can represent those who may not themselves be able to act as co-researchers, such as those with severe disabilities or young children. As such, parents play a key role in fields such as paediatric long-term illness and rehabilitation, in addition to health promotion and public health-related areas. 5 Parenting children with on-going health-care concerns presents unique challenges typically not faced by other populations due to the long-term nature of the conditions, resulting in extensive knowledge about health-care systems, child symptoms, treatment processes and more. As a result, parents may draw from very different experiences, present unique skills and have specific needs-for example, psychosocial supports due to the impact of long-term conditions on parenting-when compared with other populations. [11][12][13] How to optimally engage parents in research therefore requires further attention.
Although the concept is known by many names, this article focuses upon engaging parents as "co-researchers", stemming from the definition of user involvement employed by INVOLVE, a UK governmentfunded advisory group that supports active public involvement in health and social care research. The term refers to the act of carrying out research with/by health service users and family members who are not professional researchers, instead of to/about/for them. 7 When engaged as co-researchers, family members retain an active role and significant control over the course of the research, as well as a collaborative and interdependent relationship with the professional researchers. [14][15][16] Engaging parents as co-researchers recognizes them as experts with unique experiences and knowledge to contribute. This is believed to increase the quality and relevance of the research, 7,8 ensure acceptable and appropriate research designs, [6][7][8][9] and result in more credible and relevant outcomes. 10,17 Engaging parents as co-researchers also aligns with widespread democratic and ethical principles by allowing those affected by a health issue to influence and have a voice in the research conducted regarding such health issues and conditions. 7,9 However, despite the increasing interest in engaging parents as coresearchers, 16,18 explicit guidance on the process-how to actually do it-remains sparse. 10 Therefore, the objectives of this scoping review were to (i) synthesize what is known about engaging parents as coresearchers in health research; (ii) identify the potential benefits and challenges of engaging parent co-researchers; and (iii) map existing evidence and identify gaps in the literature. To our knowledge, this review will be the first to conduct a rigorous synthesis in this area and is the first step to developing best practices in parent co-researcher engagement.

| Methodology
We utilized scoping review methodology as outlined by Arksey and O'Malley, 19 and updated by others. 20,21 Designed to identify and map relevant literature, scoping reviews are ideal to investigate the breadth and depth of an emerging field of evidence where the literature is too heterogeneous to conduct a systematic review. [19][20][21] As such, scoping review methodology presents an ideal method for mapping, extracting and summarizing an unclear body of evidence 21 (such as engaging parents as co-researchers), in a potentially comprehensive manner. 20

| Steering committee
Scoping reviews may refer to stakeholder consultation for feedback during any or many points in the course of the study. 21 A steering committee composed of family-centred care specialists (who are also parents of children with disabilities), researchers and health-care managers collaborated throughout the course of our review. All members held an active role and were engaged throughout the course of the study, for example formulating research questions, priorities and inclusion and exclusion criteria, and providing input on article inclusion, potential resources and dissemination decisions.

| Search strategy
We conducted a comprehensive literature search across four electronic databases (CINAHL, Medline, PsychInfo and Embase) with the support of an experienced academic librarian. The search strategy for each database comprised a combination of terms representing three concepts: (i) co-researchers and research engagement (e.g. family engagement, parent involvement), (ii) the population engaged (e.g. parent, family) and (iii) the field of focus (e.g. research, biomedical research). The search was limited to articles published from 2005 to 2015, and in English. See Appendix 1 for a sample search strategy.
Hand-searching was also conducted with reference lists of included articles, as well as selected relevant bibliographies. 22,23 Several sources of grey literature were reviewed (listed in Appendix 2). Content experts on our steering group were also consulted for potential resources and sources of grey literature. A full list of search strategies, subject headings and keywords can be obtained from the last author.

| Inclusion/exclusion criteria
To be included in the review, articles had to (i) focus on engaging parents/guardians (herein parents) in the process and/or design of research; (ii) describe the process and/or benefits of engaging parents in conducting research; and (iii) be related to health care. Exclusion criteria included the following: (i) parents were solely research subjects/ participants; (ii) research focused solely on mental health, sexual health or oral health, given the specialized nature of these areas that warrant a separate review. Articles were also excluded if they engaged solely first-time parents immediately post-partum due to lack of experience in the parental role. Additionally, articles that provided insufficient data to complete 50% of the key fields in our data extraction table were excluded.

| Data selection and extraction
Articles were initially screened using the inclusion and exclusion criteria via title and abstract. One reviewer (AS) screened all abstracts (n=8994), and a second researcher reviewed approximately 100 abstracts to calculate inter-rater reliability. Conflicts were resolved by consensus, and a third reviewer (ACM) was brought in to review and settle further discrepancies. As inter-rater reliability was high, author AS continued as the primary reviewer, retrieving and screening full text articles. Using the inclusion and exclusion criteria listed above, the primary reviewer (AS) was able to reduce all search results to the final included articles. All full text articles to be included were reviewed by the steering committee for relevance then charted in an electronic database created with Microsoft Excel via an iterative process concurrent with data extraction. 21 The final table was checked by ACM for accuracy.

| Summation, collation and synthesis
To map the available literature and identify significant gaps, 19 an initial numerical summary was completed to provide an overview of included studies' characteristics, such as the types of research design, geographical location, year of publication and study populations. Themes drawn from the papers that were relevant to the objectives were identified from the extracted data, grouped and discussed by the steering committee.

| Quality assessment
While study inclusion is not determined by quality in scoping reviews, 19

| RESULTS
Our initial search yielded over 11 000 articles; 8994 were retained after removing duplicates with reference management software.
Nine articles were included in the review. Inter-rater reliability in the abstract screening phase averaged 99.02%. After hand-searching and grey literature review, an additional report was identified, resulting in a total of 10 documents (herein referred to as "articles" for ease) that matched our inclusion criteria (Table 1). A full overview of the search process can be found in Fig. 1.

| Article characteristics
The articles were published in the United Kingdom (n=5), United States (n=4) and the Republic of Ireland (n=1). The majority (n=6) of articles were published in or after 2009. Little detail was provided regarding the characteristics of the parent co-researchers. Selected characteristics regarding the population and research settings among included articles can be found in Table 2, which highlights patterns in demographic data, number of parent co-researchers and the settings, when specified.

| Terminology used
A wide range of terms were used across studies to describe coresearcher engagement. Most common were as follows: "patient/ public/user involvement" (n=3) and "participatory research" (n=3), followed by "co-researcher" (n=2). The terms "parent co-investigator," "parent researcher," "lay researcher," "participatory action research," "community-based participatory research" and "community-based participatory research" were each used in a single article.

| Design and quality
Included articles were classified as case studies (n=6), descriptive articles (n=3) and reports (n=1). Healthy lifestyles and obesity research was the most common field of study (n=4), followed by disability and long-term disease (n=3), and infant health (n=3). The majority of articles engaged parent co-researchers in research informing programme development, implementation and/or evaluation (n=6). Quality assessment was applicable to six empirical articles, with a mean score of 39.2%. The maximum score was 45.2%, with a lowest score of 31.0%.

| Recruitment and group composition
The majority of the articles (n=6) 25-30 made use of existing relationships (e.g. contacting members of a hospital advisory group or community programme) to identify potential parent co-researchers. Other recruitment methods included the use of advertisements or job postings, 26,[29][30][31] or leveraging key community contacts such as nurses or teachers. 29,32 Most researchers engaged parent co-researchers continuously through the course of the project via either small groups of the same one to four parents, 25,29,30 or large groups with fluctuating member composition. 26,28,31,32 Other researchers made use of large focus groups of parents for input at intermittent points through the course of the research process. 25

| Research processes
Studies involved parent co-researchers in different aspects of the research process (Table 3 provides an overview). Parent coresearchers were reported as having been engaged in developing research questions, aims and objectives and setting research priorities. 25

| Data analysis and dissemination
After the collection of data, several articles stated that parent co-researchers performed data entry and analysis 25,26,28,29,32,34 and formulated recommendations. 25

| Evaluating success of parental engagement
When specified, articles reported using anecdotal comments to evaluate the result of parent co-researcher involvement, 25 Three of the articles reported that the researchers analysed the proceedings of the research project to determine results via analysis of recordings and self-reflections to identify key themes. 26,30,31 In one study, the impact of involving parent co-researchers in making messages more relevant and meaningful to other parents was assessed by comparing the rates of parents recalling having seen the campaign messages to those "typically observed in child health promotion campaigns targeting parents through mass media." 34 p31 Three articles did not mention the use of any form of outcome evaluation. 27,29,33

| Benefits for researchers
Nine of the 10 articles concluded that parent engagement led to more sustainable and population-appropriate interventions. [25][26][27][29][30][31][32][33][34] The resulting research was reported to be more meaningful and culturally/ socio-economically appropriate, identifying issues and details that researchers may not have been initially aware of. For example, parent co-researchers helped to optimize intervention timing and the location of data collection to accommodate the needs of participants, thereby maximizing participant involvement and attendance, and thus increasing the amount of data gathered (e.g. response rates on a survey). In addition, four articles made note of the passion and enthusiasm exhibited by parent co-researchers, which motivated researchers and fuelled momentum and ideas for future research. [25][26][27]30 Four articles identified wide-ranging experiences between parent co-researchers, in both their experiences of caring for children with differing needs, and in how their lifestyles differed. 25,29,31,33 These factors were thought to have contributed to an increased pool of expertise and opinions, leading to greater rigour in decision making and overall increased quality of results. Parent co-researcher involvement was also thought to have increased the reach of dissemination, 25,27,33 as well as the credibility of the research with patient/parent groups and professionals. 26,27,29,30

| Benefits for parents
Several articles reported parent co-researcher empowerment resulting from engagement in the research, due to increased confidence and research skills, 26,30,32 as well as obtaining a sense of control over health service involvement. 25,27 Engaging parent co-researchers also increased their awareness of health issues and increased the likelihood of making changes in the area of focus in the future, 27  incorporate their feedback. 25,26,29,33 Increased monetary costs could also be incurred as a result of more time spent on the project, and the need to compensate and support the parent co-researchers.
A further challenge arose from disconnects between parent co-researcher and researcher foci of interest, as researchers were perceived to be driven by literature gaps and funding agency directives, which did not always align with the concerns of the parent co-researchers. [25][26][27] Two articles addressed the risk of a power imbalance between researchers and parent co-researchers, reporting that researchers should take a facilitative, rather than leading role, as per the traditional research paradigm. 26,27 For example, researchers would often lead the actual data analysis and/or relay information to parents in a synthesized and lay manner. [25][26][27]30,32 Several studies raised concerns that the true power still remained with the researchers, potentially resulting in tokenistic parental involvement. 26,31 As a result, a number of articles addressed the need for researchers to have specialized skills to truly engage parents as equals. 25-27,31,33

| Challenges for parents
Differences among parent co-researchers, and/ or between parent co-researchers and researchers appeared to create several challenges.
Inconsistent educational levels and research expertise were reported to result in parent co-researcher disappointment, frustration and powerlessness due to a lack of awareness of certain research logistics, for example the inherent unpredictability of methods and results in research, [25][26][27]29 as well as the lack of immediate action as a result of their contributions. 26,27,30 Two articles also outlined potential parent disengagement resulting from unclear roles and task distinctions between the researchers and parent co-researchers. 29 It is important to note that a substantial body of literature already exists on the topic of patient engagement, including descriptive articles and frameworks, 5,6,9,15,35 case studies, 14,17,36-38 reports 2,7,16,39 and reviews. 8,10,18,[40][41][42]  Even where researchers are willing to engage with parent coresearchers, we did not identify any clear guidelines on how to do so.
However, the results of this scoping review provide some indication of practices that may be beneficial and can serve as the basis for subsequent research endeavours to inform best practices when researchers are engaging parents as co-researchers (see Box 1 for an overview).
One key challenge when conducting this scoping review was that the concept of engaging patients and families has been referred in the literature using diverse terminology. Among the articles included in this review, no single term had more than three articles using it, with the majority of terms having only one instance. Within the broader literature, Turnbull et al. have identified the terms "participatory research," "action research," "participatory action research," "constituency-oriented research and dissemination", "emancipatory research", "empowerment research", and "discovery research". 9 Additionally, the terms "patient/public involvement," "patient/ service user engagement," "lay involvement" and "public consultation" Definitions were also rarely provided, taking additional time to carefully determine the level of parental involvement in the research project. The terms "participatory action research," "action research" and "emancipatory research" are considered a qualitative research method engaging community members and settings, 46 but are conceptually distinct from parental co-researcher engagement that we are examining (which is an element that can be integrated into any project). 7 However, some papers conflated the terms and used "participatory action research" alongside elements of co-researcher engagement. It was therefore challenging to distinguish between different approaches and reinforces our call for standardized terminology to be used. We therefore recommend having a universally defined set of terminology to avoid such challenges and advance future research. "Patient engagement" has become commonly used, including with the U.S. setting and public service campaigns. 8,10,49 However, as the term is still somewhat ambiguous, "co-researcher" may be appropriate as an additional, more categorical term to refer to patient engagement in research specifically.

| Considerations
The research included in this study was primarily conducted in the United Kingdom and United States, reducing the generalizability of their recommendations (where stated). There is therefore a clear lack of evidence across different countries, which is potentially important due to differences in health-care delivery and research settings.
Due to the emerging state of the evidence, the full impact of parent co-researchers on the research field is as yet unclear. However, as parents remain proxies for the voice of children with severe disabilities for most or the entirety of their lives, there are clearly potential benefits to exploring further how best to engage them in research.
Although scoping reviews are not designed to give weight to studies' findings based on quality assessment, 19,21 the diversity in the quality of studies that we located must be considered when interpreting our findings. For example, the six empirical studies that underwent quality assessment were of relatively low quality, with the highest quality score being 45.2%, and the lowest being 31.0%. One reason was the lack of any specific framework, resulting in inconsistencies and lack of structure among the studies. There was a notable lack of demographic data in the included articles, such as the age of parent for full text screening. This is one reason why we screened 101 full text articles, plus additional hand-searching, but only yielded 10 final articles.

BOX 1 Recommendations for engaging parents in research
• Engage parents as early as possible to build relationships to maximize their impact 7,8,17,25,27,29,32,36 • Provide support, encouragement and recognition to parents for their contributions, recognize parents as experts and equals, 27,28,31 be responsive to parents' lifestyles. 25,29 Some of the topics discussed are very personal to the parents and can elicit emotional responses 7,8,15,[25][26][27] • Be clear on roles-outline the differences in duties and expectations between the co-researchers and professional

| CONCLUSIONS
The current evidence suggests that engaging parents as coresearchers brings both benefits and challenges, but can potentially enhance research that is acceptable and relevant for the population it is intended to serve. Despite this, there is insufficient high-quality research to create evidence-based best practice guidelines for how to engage parents as co-researchers at this time. However, our scoping review provides a synthesis of available evidence on this under-researched topic and can guide future research focusing upon structured frameworks and rigorous approaches to engaging parents equitably in health research.