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INTRODUCTION: The importance of including people affected by research (e.g., community members, citizens or patient partners) is increasingly recognized across the breadth of institutions involved in connecting research with action. Yet, the increasing rhetoric of inclusion remains situated in research systems that tend to reward traditional dissemination and uphold power dynamics in ways that centre particular (privileged) voices over others. In research explicitly interested in doing research with those most affected by the issue or outcomes, research teams need to know how to advance meaningful inclusion. This study focused on listening to voices often excluded from research processes to understand what meaningful inclusion looks and feels like, and asked what contributes to being or feeling tokenized. METHODS: In this deliberative dialogue study, 16 participants with experience of navigating social exclusions and contributing to research activities reflected on what makes for meaningful experiences of inclusion. Using a co-production approach, with a diversely representative research team of 15 that included patient and community partners, we used critically reflective dialogue to guide an inclusive process to study design and implementation, from conceptualization of research questions through to writing. RESULTS: We heard that: research practices, partnerships and systems all contribute to experiences of inclusion or exclusion; the insufficiency or absence of standards for accountability amplifies the experience of exclusion; and inclusive practices require intention, planning, reflection and resources. CONCLUSIONS: We offer evidence-informed recommendations for the deeply relational work and practices for inclusivity, focused on promising practices for cultivating welcoming systems, spaces and relationships. PATIENT OR PUBLIC CONTRIBUTION: This work reflects a co-production approach, where people who use and are affected by research results actively partnered in the research process, including study design, data-generating activities, analysis and interpretation, and writing. Several of these partners are authors of this manuscript.
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BACKGROUND: In 2017, the British Columbia (Canada) SUPPORT (SUpport for People and Patient-Oriented Research) Unit created six methods clusters to advance methodologies in patient and public oriented research (POR). The knowledge translation (KT)/implementation science methods cluster identified that although there was guidance about how to involve patients and public members in POR research generally, little was known about how best to involve patients and public members on teams specifically exploring POR KT/implementation science methodologies. The purpose of this self-study was to explore what it means to engage patients and the public in studies of POR methods through the reflections of members of five KT/implementation science teams. METHODS: Informed by a collaborative action research approach, this quality improvement self-study focused on reflection within four KT/implementation science research teams in 2020-2021. The self-study included two rounds of individual interviews with 18 members across four teams. Qualitative data were analyzed using a thematic analysis approach followed by a structured discussion of preliminary findings with the research teams. Subsequently, through two small group discussion sessions, the patients/public members from the teams refined the findings. RESULTS: Undertaking research on POR KT/implementation science methodologies typically requires teams to work with the uncertainty of exploratory and processual research approaches, make good matches between patients/public members and the team, work intentionally yet flexibly, and be attuned to the external context and its influences on the team. POR methodological research teams need to consider that patients/public members bring their life experiences and world views to the research project. They become researchers in their own right. Individual and team reflection allows teams to become aware of team needs, acknowledge team members' vulnerabilities, gain greater sensitivity, and enhance communication. CONCLUSIONS: The iterative self-study process provided research team members with opportunities for reflection and new understanding. Working with patients/public team members as co-researchers opens up new ways of understanding important aspects of research methodologies, which may influence future KT/implementation science research approaches.
In this self-study we aimed to understand how members of research teams work together. We explored how research teams included patients and public members in studying research approaches (methods) that support the creation, sharing, and use of research results that matter to patients and the public (patient and public-oriented research). We analyzed interviews with 18 members of 4 teams and discussed the findings with each team. We then further refined the results and their interpretation with patients/public team members. We found that research teams had to deal with the uncertainty that is common for this type of research, which is often exploratory and focuses on processes. There needed to be a good match between patient/public members, the topic, and the team. Teams had to be flexible in how they worked together and they needed to address how power was shared within the team. Finally, the teams were affected by world events such as COVID and social unrest. Research team members had a chance to reflect and gain new understandings through the self-study. A key learning was that patients and public members bring their life experiences and world views to the project. They become researchers in their own right. When team members have a chance to reflect they can become more aware of their own and others' talents, needs, and vulnerabilities, that awareness can help improve communication and teamwork. Patients and public members as co-researchers can bring new ways of understanding important features of knowledge translation and implementation science research approaches.
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Fundamental processes influencing human growth can be revealed by studying extreme short stature. Using genetic linkage analysis, we find that biallelic loss-of-function mutations in the centrosomal pericentrin (PCNT) gene on chromosome 21q22.3 cause microcephalic osteodysplastic primordial dwarfism type II (MOPD II) in 25 patients. Adults with this rare inherited condition have an average height of 100 centimeters and a brain size comparable to that of a 3-month-old baby, but are of near-normal intelligence. Absence of PCNT results in disorganized mitotic spindles and missegregation of chromosomes. Mutations in related genes are known to cause primary microcephaly (MCPH1, CDK5RAP2, ASPM, and CENPJ).
