Improving social justice in observational studies: protocol for the development of a global and Indigenous STROBE-equity reporting guideline

Our overall goal is to develop guidance on reporting health equity considerations in observational studies to make health equity data more readily available for synthesis and decision-making.

This goal has two distinct components, which are:

We recognize that the harms of colonization have affected Indigenous Peoples globally. However, we decided to focus on these four countries since they have a similar experience of colonization which included land dispossession and policies for assimilation [55]. We sought Indigenous scholars from each of these countries to join our Indigenous Steering Committee and designed methods to collect data on research methods in these countries (see below). We considered that this project did not have sufficient resources to adequately appraise the empirical data and seek sufficient global representation to develop reporting guidelines for all global Indigenous Peoples. By transparently reporting our methods and processes, we hope this guideline may be helpful for adaptation for other Indigenous Peoples in different global contexts. Indigenous Peoples, scholars, public members will be given opportunities to review the global consensus guidelines as well. We feel the entire project benefits from Indigenous perspectives.

The research to achieve this goal will be interconnected and empowering, with opportunities for reciprocal learning through joint meetings and communication. Indigenous approaches to research include taking a strengths-based, anti-oppressive and anti-colonial approach [56], founded on authentic, ethical partnerships [57, 58] and following principles of Indigenous data sovereignty [59]. These approaches are central to understanding health inequities and social justice in designing, conducting, and reporting research.

This project aims to improve the consistency of reporting of health equity in observational studies, by fulfilling the following specific objectives over the span of 4 years:

The study will comprise five phases, adapted from guidance for developing reporting guidelines by Moher et al. [60], including establishing the need for guidance, seeking comprehensive feedback, establishing consensus, evaluating outcomes of learning across streams and implementation over the span of 4 years. Members of our team have adopted this approach for other reporting guidelines on equity in systematic reviews [61, 62] and randomized trials [CONSORT-Equity] [63]. The five phases of the current study are: (1) Assess available guidance, viewpoints, and reporting of health equity in observational studies, (2) Seek wide international input using a survey on potential items to improve reporting of health equity, (3) Establish consensus amongst knowledge users and researchers on the health equity reporting guideline, (4) Evaluate the relevance of the reporting guideline for Indigenous research, and, (5) Widely disseminate and encourage uptake of STROBE-Equity (Fig. 1). In our study, we focus on Indigenous Peoples in Australia, Canada, New Zealand and the USA given the similarity in colonization experience and representation on the Indigenous Steering Committee for this project, as described above.

We will review the candidate items supported from scoping review and the items found to be poorly reported in the methods study. Study design details regarding the methods studies will be published on open access platforms or published in peer review journals when possible [64‐66]. We will analyze focus groups thematically and use themes to generate additional candidate items. All the items will be complied and synthesized to develop a final list of items to be presented in an online survey. The survey will be used to gather international feedback on the suggested list of items and identify gaps where new items might be required. The results of the survey will be analysed quantitively by calculating the frequencies and percentages of each response option for each question. We will assess the agreement on the candidate items and hold the consensus meeting to discuss the final list of items. Subsequently, we will develop a writing team to wordcraft and refine the items in response to feedback on drafts, and compile examples identified by the empirical studies and expertise of the team.

We will conduct this work in parallel to the stream focused on Indigenous research led by our Indigenous principal investigator (SF), with engagement of relevant Indigenous knowledge holders and knowledge users and will follow the same steps. The two streams of research will be interconnected and empowering with opportunities for joint learning through regular communication. To articulate the connections between the two streams of research, we consulted with an Indigenous artist Claire Brascoupé (Algonquin Anishinaabe artist) to design a figure of our shared vision for parallel streams, with interconnected elements (Fig. 2). The process of co-designing this figure with our steering committees helped to establish our methods for enabling separate streams with multiple points for connection and learning from each other.

We will conduct six linked empirical studies on how health equity is reported in observational studies; three focused on global research and three on research on Indigenous people. We define observational studies according to those relevant to the STROBE reporting guideline as case-control, cohort, and cross-sectional studies, which collect and report quantitative data, e.g., studies conducted using administrative data, natural experiments, and prospective cohorts.

We will use an organizing framework to document evidence about health inequities defined by the acronym: PROGRESS-Plus, which stands for Place of residence (urban/rural), Race/ethnicity/culture/language, Occupation, Gender or sex, Religion, Education, Socioeconomic status and Social capital [11]. The “Plus” characteristics are other characteristics associated with health inequities such as age, disability, sexual orientation, and temporary exposure to vulnerable circumstances such as discharge from hospital and food security [67]. We will apply this PROGRESS-Plus lens to our global studies as well as to our studies focused on observational studies with, for and about Indigenous peoples.

We will develop an online survey of potential items by triangulating, synthesizing and integrating findings from each of the studies conducted in Phase 1 (survey findings the scoping review, the methods review, the key informant interviews) to develop good practice examples. The steering committees and Technical Oversight will be consulted for feedback, and the draft survey will be tested with potential respondents. Our Technical Oversight and steering committees will be asked to identify at least 200 global individuals to participate in an online survey using Survey Monkey (https://​www.​surveymonkey.​com/​) on the importance of potential items and will be invited to provide open-ended comments about each item. The participants will represent different intended knowledge users, researchers who conduct observational studies, ethics board representatives, journal editors, funders, decision-makers, and patients/public. Similarly with other aspects of our project, we will seek diverse respondents to include people with experience across all aspects of PROGRESS-Plus, geographic diversity, age, gender, and country income balance. We will collect details on sociodemographic characteristics and disciplinary background. For patients/public, we will conduct face-to-face video or phone interviews according to their preference. We have used this approach successfully for other guidelines, achieving over 200 responses across diverse collaborators [63, 79]. We will use the CHERRIES checklist for reporting the survey results quantitatively [80], including tables and figures.

We will carry out a separate survey of Indigenous knowledge holders, decision-makers, and the public, and these results will be reported according to CHERRIES and summarized with descriptive statistics in tables and figures.

For the global guidance, we will hold a consensus meeting of 25-30 international, multidisciplinary and diverse collaborators to develop the STROBE-Equity extension. The Technical Oversight and steering committees will suggest people according to the following criteria: opinion leaders, standard setters, and experts in disciplines relevant to observational studies and equity (e.g., methodology, social sciences, public health, health equity), intergovernmental organizations and multilateral agencies, collaborators with diverse perspectives (e.g., funders, editors, practitioners) and patients with lived experience of health inequity. We will consider balance in participants across characteristics such as career stage/age, gender and low- and middle-income countries (LMIC)/high-income countries (HIC). At least 10% of participants will be people with lived experience, and at least 20% will be Indigenous. The number of participants is based on our experience of similar meetings as the maximum to allow meaningful discussion and is similar to the number recommended by other organizations such as the James Lind Alliance [81]. Two weeks prior to the meeting, participants will be given pre-reading materials, including results of empirical studies. To ensure sufficient time, we will hold a pre-meeting vote by all invited participants to identify areas where more discussion is needed. We will summarize evidence from the above studies (conducted in Phase 1) and exemplars of best practices for each item. We have found this promotes discussion. For each item, we will seek consensus using the Nominal Group Technique [82]. We will discuss and decide whether to publish a single manuscript on the STROBE-Equity extension or develop a short Statement paper, and a separate Elaboration paper with examples, e.g., Preferred Reporting Items for Systematic reviews and Meta-Analyses extension for Equity (PRISMA-Equity) was published as two papers [61, 62]. A writing team will be formed to develop the manuscript(s). These will be refined through iterative discussion with all participants, steering committees, and Technical Oversight members by email, face-to-face meetings, and videoconference calls. Discussion will be documented regarding the rationale for each item.

We will conduct a separate consensus meeting for guidance on reporting health equity in Indigenous research in Australia, Canada, New Zealand and USA. This meeting will be led by Indigenous peoples and will include Indigenous researchers, knowledge holders such as community elders, decision-makers, and patients/public from Australia, Canada, New Zealand and USA. The participants will be determined in collaboration with our Indigenous research steering committee. The approach will be based on community-based participatory research and Ownership, Control, Access and Possession (OCAP®) [83] and Collective Benefit, Authority to Control, Responsibility, Ethics (CARE) [84] principles. We approach this research from a relational perspective and will focus on communities and populations who are represented on the Indigenous steering committee.

The focus on Australia, Canada, New Zealand and the USA may limit applicability and relevance of this reporting guideline for Indigenous Peoples in different countries with different colonization experiences. However, we hope that our approach and results may be useful for adapting or adopting this guidance for other global Indigenous Peoples, with leadership from Indigenous Peoples in those countries.

We plan to explicitly document the outcomes of collaboration between the parallel stream of the global guidance and the Indigenous stream focussed on Australia, Canada, New Zealand and USA. As indicated in Fig. 1, we plan to share methods and processes developed within each stream. We anticipate that there may be innovations developed in one stream than can inform the other. Documenting these examples may be helpful in designing future studies with parallel processes.

After discussing with the journal editors involved, we plan to publish the STROBE-Equity extension as a pre-print to allow early access and feedback prior to publication. This will allow feedback from a wider audience. Also, it will allow us to begin the end of grant knowledge translation activities soon after the consensus meeting when the participants are actively engaged. As mentioned earlier, the lack of implementation tools has been described as a barrier to the uptake of reporting guidelines [85]. Thus, we will develop practical tools (e.g., checklists for journal editors and short podcasts or stories for the public) based on consultations with the steering committee that include journal editors, and we will promote these through our connections with journal editors.

While there is no systematic strategy to disseminate reporting guidelines, we will disseminate the products to a wide range of global and targeted audiences through publications and presentations, and dialogues and exchange activities with knowledge user groups. We have registered this planned reporting guideline with EQUATOR (http://​www.​equator-network.​org/​). We host a web page on the Campbell and Cochrane Equity Methods Group website (https://​methods.​cochrane.​org/​equity/​) where we will post publications, and links to surveys, tweets, and other news. Our collaborators will also be asked to suggest ways to reach their communities, for instance, through dialogues and face-to-face sessions.

We will develop an evaluation framework to assess these activities using indicators such as LinkedIn and Twitter activity (numbers of tweets, retweets, likes, and reach), invited presentations (online or face to face), number of people reached by presentations, news articles and outlets, Facebook activity (likes, shares), LinkedIn likes and replications, citations of our articles or blogs in journal articles, or presentations.

In developing this project, we first identified common interests in health equity in observational studies amongst our international network of patients, practitioners, and researchers with expertise in systematic reviews and scoping reviews, observational studies, health equity and knowledge translation. We established three steering committees: one for Indigenous research, one for knowledge users, and one for any other members of the public. We set up a governance structure of an executive team of four principal investigators (PIs) and a lead for each of three steering committees and a Technical Oversight committee to guide how we would work together. This team has expertise in statistics, social science, epidemiology, Indigenous health, knowledge translation, equity, reporting guidelines, lived experience of inequities, and using evidence to inform decisions. We defined terms of reference based on our prior experience, including principles of equitable partnership [86, 87]. We have co-developed an inclusive governance plan to support our plan to co-produce this research through a partnership that values different types of knowledge and participation [86, 88] (Fig. 3).

The executive team of four Principal Investigators (VW, LM, JJ, SF), a research coordinator and a post-doctoral fellow will meet regularly to consult on study methods and issues that arise during the conduct and provide quarterly updates to the whole team. The executive team will meet quarterly by video conference with the Technical Oversight Committee and the Indigenous research, patient/public and knowledge user steering committees.

Co-investigators will be invited to sign up online for all studies/papers they are interested in co-authoring. Also, we will use online tools to enable the informal exchange of ideas amongst co-investigators. Authorship for papers will be governed by terms of reference (76, 87) (Additional file 2).

Coinvestigators will participate in quarterly Technical Oversight calls for strategic direction, study design planning with their expertise in Indigenous health research. Graduate trainees will lead or co-lead empirical studies with mentorship from senior investigators.

We have developed a patient and public steering committee inclusive of diverse populations with an interest in health equity across dimensions of PROGRESS-Plus co-led by patient and patient representatives. The representatives have been engaged since the development of the project and will convene bimonthly to advise on the design and conduct of the studies, and the interpretation of the data. We will seek diverse viewpoints based on lived experience and values [89] and evaluate experiences of engagement using available tools [90, 91].

This 4-year project has been developed using an integrated knowledge translation approach with a diverse, global multidisciplinary team, including patients, authors of observational studies, statisticians, social scientists, epidemiologists, methodologists, funders, ethicists and knowledge users [86]. Our team is diverse and representative of LMIC and HIC, gender, ethnicity, career stage and disciplines. We have assembled knowledge users from (PAHO/WHO), the Royal College of Physicians and Surgeons of Canada, journal editors, the Canadian Agency for Drugs and Technologies in Health, the Public Health Agency of Canada. We will design an evaluation framework to assess the process and outcomes of this approach, including the experiences of all involved, led by JJ [87].

We will use twitter (e.g., @cochraneequity, @vawelch, @EquityStrobe), mailing lists and other fora to seek input from external collaborators. Our geographic and disciplinary diversity will help us seek diverse opinions and reach a broad audience. We will develop a one-page policy brief lay summary of each deliverable aimed at government decision-makers and journal editors. We will develop digital stories to engage the public and patients [92]. Accessibility of results will be enhanced by using reading level assessments for our policy summaries, blogs, surveys and presentations. We will translate the results into Portuguese, French and Spanish. Other translations will be encouraged (e.g., Chinese) as conducted for the CONSORT and original STROBE guidelines (https://​www.​equator-network.​org/​library/​translations-of-reporting-guidelines).

A strength of our approach is the development of a parallel stream on Indigenous research which may be used as a model for creating guidance for other specific populations with different lived experience of health inequities. This requires ethical processes to facilitate culturally competent research that respects Indigenous values and ensures space for culturally safe dialogue [93]. To address this, one of our co-PIs is an Indigenous researcher with training in Ownership, Control, Access and Possession (OCAP^(R)) [83]. Our Co-PI responsible for leading the development and evaluation of integrated KT is also trained in OCAP^(R) and has experience working with Indigenous communities [93, 94]. We have developed an Indigenous Steering Committee with Indigenous researchers from Australia, Canada, New Zealand and the USA to bring a high-income country and shared British colonial experience perspective. We will follow the principles of the United Nations Declaration on the Rights of Indigenous Peoples (UNDRIP) [19].

We will ensure regular communication with videoconference software, with screen share capabilities, thus minimizing environmental impact. Minutes will be distributed to allow all team members the opportunity to participate. One-on-one videoconferencing meetings (or small groups) will be used to seek specific expertise where needed.

Observational studies include diverse studies from cross-sectional surveys and natural experiments to analyses of administrative health records and longitudinal cohorts. The feasibility of taking on this broad range of studies is increased by having members of the original STROBE statement. The team includes global leaders in non-randomized studies from the Cochrane and Campbell Collaborations [95] and expertise in consensus methods [96].

A strength of our approach is the use of different methods to seek input and perspectives on the proposed items, which include an in depth key informant qualitative study, a global survey (phase 2) and a consensus process which integrates the empirical findings and survey findings with a diverse group of stakeholders. These three ways to integrate external perspectives aim to bring global perspectives to the work.

Poor implementation and lack of integration with policies, plans and the structure of research publication and knowledge management (e.g., international research registry data providers) has hindered the impact of prior STROBE guidelines [97‐99]. To mitigate this risk, we have planned for 1 year of knowledge translation activities after the consensus meeting and resources for end-of-grant implementation activities, including the development, implementation, and evaluation of tools for authors, sponsors, standard setters, and editors.

Given the widespread recognition of the need to redress systemic racism, sexism and all forms of discrimination in medicine [100], education [101], and justice [18], these reporting guidelines have the potential to expand awareness about the need to consider health equity in observational studies change and reporting practices on health equity in observational studies, which comprise the majority of health research. In turn, uptake and use of this guideline can substantially add to our knowledge about which health inequities are prevalent and how to redress them both within and between countries. A parallel stream focused on research with Indigenous People is a novel aspect of this guideline development, and the approach could be modelled for research with other populations who face systemic discrimination, such as people living with disabilities. We believe this will subsequently improve the evidence base on health equity in observational studies to improve social justice in policy and practice decisions.