This study examined the characteristics of randomized trials and patient priorities for exercise science research for people with T1D. In our scoping review, we determined that patient engagement methods and PROs have not been historically used to inform exercise-based interventions. Guided by these results, we facilitated a priority-setting project with T1D stakeholders to identify priority research questions pertaining to exercise and health. We identified that patients and caregivers are interested in modalities and strategies to exercise safely and maintain glucose control. Collectively, these findings provide a novel patient-centered rationale for designing future randomized trials of exercise interventions for people with T1D.
Previous literature
This is the first scoping review of exercise randomized trials for individuals with T1D designed to determine if patient engagement exists in exercise and T1D literature. This topic was not addressed in recent systematic reviews of exercise training and health outcomes in people with T1D.41–43 We found that exercise randomized trials published or being delivered for individuals with T1D did not focus on stakeholder engagement. This gap is not exclusive to trials of T1D and exercise. A scoping review of priority-setting practices in all health research found only 27 studies engaged patients in identifying research topics, and 12 in identifying specific research questions.44 Many studies simply inferred stakeholder priorities from qualitative data. Additionally, most trials engaging stakeholders do not integrate multiple stakeholders’ perspectives (ie, patients, clinicians, caregivers, etc) in the prioritization process. This is an important consideration when engaging stakeholders in research, as stakeholders with different experiences of a health condition may have different priorities for research topics or outcomes.45 Engaging T1D stakeholders is a significant gap in exercise science literature and should be considered within future randomized trials.
As of yet, PRO reporting has been minimal across T1D and exercise randomized controlled trials. This may be due to the lack of agreement on a core list of PROs to be measured in this field. Unfortunately, the core list of PROs determined by the D1 Now study in Ireland12 includes outcomes that are not relevant to exercise interventions (eg, level of clinic engagement). It may be prudent for researchers and stakeholders to co-produce a core list of PROs for T1D and exercise in the near future. Some exercise trials have measured PROs as primary or secondary outcomes,31 35 46 a practice we hope to see translated into more randomized controlled trial designs as well. However, it is important to recognize that PRO measurement may not be suitable for all exercise trials, particularly those with small sample sizes, and qualitative interviews investigating intervention acceptability may be more appropriate.
The stakeholders engaged as steering committee members were recruited to reflect variance across age, sex, ethnicity and rural versus urban residence categories. These four variables were identified from previous studies that used these sampling methods47–49 and were selected by consensus from five members of the research team (NMDK, SDM, KMS, TAD and JMMG). It is important to recognize that other social determinants outside of these selected variables could have impacted the resulting list of prioritized questions (ie, household income, religion, duration of diabetes, etc). However, in keeping with recommendations from the JLA regarding steering committee size, only a select number of factors could be used in recruitment to adequately reflect differences within each. Despite the inability to recruit steering committee members across additional social determinants, our survey sample spanning nine provinces in Canada provides some confidence in the representativeness of the resulting priorities.
As with any research involving group-based activities, it was important for our team to consider the possible effects of perceived power dynamics in small group discussions and ranking exercises. This was particularly important since youth and multiple stakeholder types (both patients and healthcare providers) were involved in this project. As per the JLA guidebook,14 an adapted nominal group technique50 guided the final workshop procedures. This permitted anonymous voting on individual’s level of agreement with aggregated lists in between each small group discussion session, to ameliorate the risk of individuals simply agreeing with decisions made by fellow group members. Additionally, a research team member was assigned to each small group (NB, JLH, JMMG) during discussions as a moderator to support fair engagement among all group members. The research coordinator (NMDK) floated between all groups to help facilitate discussions and affirm rankings were reflective of stakeholder discussions.
Patient engagement and priority-setting projects identifying important research topics from stakeholders’ perspectives are becoming more common within clinical research.44 51 52 This project revealed that stakeholders are largely concerned with short-term outcomes, strategies to prevent hypoglycemia and stabilizing short-term glucose control. This contrasts the JLA T1D treatments project,13 where prioritized questions focused on long-term outcomes including adverse effects of various insulin analogues or potential cognitive impacts of living with T1D. This difference may indicate some uncertainty felt by stakeholders regarding the safety of exercise given their individual situation. Fear of short-term health complications is a common barrier to regular exercise among people with T1D.5 This fear itself has a range of health implications including reduced physical activity,53 increased glycemic variability,53 poorer sleep patterns54 and reduced quality of life.54 Although guidelines and consensus statements about prevention of postexercise hypoglycemia exist,55 56 the literature on which these recommendations are based has limitations. As this is the first investigation into patient priorities in T1D and exercise, previous research may not have been intentionally directed towards established patient-identified questions. Future randomized trials should focus on stakeholder priorities to provide optimally relevant recommendations to individuals with T1D.
Strengths and weaknesses
This study was strengthened by using two complimentary methods to identify and address the gap of patient priority-setting and engagement in exercise science for individuals with T1D. The scoping review approach supported a rigorous and systematic search of published and unpublished literature sources. This search strategy provided the richest base possible to analyze the narrative of previously conducted studies using a patient-oriented research lens. The narrative approach of scoping reviews enabled discussion of the existing literature beyond summaries and quantitative meta-analyses that have recently been conducted by other authors in the field.42 57 The priority-setting project followed a modified JLA approach to priority-setting, and the recognition and support for this model22 is a strength for our study. The steering committee recruited for this project were highly engaged (100% phase III participation rate), which served as assurance that a patient-oriented approach to research moving forward would be valued by this stakeholder population. Despite these strengths, several limitations should be addressed. Although scoping reviews do not require a critical analysis of included studies,16 this is not a limitation of this review as our purpose was simply to identify instances where stakeholders may have been involved in research decision-making. However, the scoping review was limited to trials published in the English language over the past two decades, therefore some trials engaging patients or other stakeholders may have been missed. Additionally, the authors recognize that reporting of patient engagement is only recently growing within the literature, although still not captured in the Consolidated Standards of Reporting Trials guidelines for reporting randomized controlled trials. As a result, patient engagement practices may have occurred but not described in every publication, particularly within journals having a lower word limit. The EXTOD (EXercise for Type One Diabetes) study serves as one example of various publications addressed different aspects of the research design and outcomes, including qualitative patient engagement practices in the early design phase.58 59 However, data for this review were extracted strictly as reported within articles. First authors were not contacted and related publications were not searched for to confirm whether patient engagement played a role within the studies. Future reviews with a similar purpose may want to explore possibilities for this additional data collection. In terms of the priority-setting project, one limitation is that consensus was not achieved in person at the final workshop. Although the JLA Guidebook14 mentions that it is not uncommon for consensus to be difficult to achieve and a majority vote can be obtained in these situations, this process had to be conducted in a postworkshop analysis since several members had other commitments.