Browsing by Author "Marshall, Deborah A."
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Item Open Access A Comprehensive Case Study of an Orthopaedic Surgery Central Intake Service in the Winnipeg Regional Health Authority: A Single-Entry Model to Manage Waiting Times for Total Joint Replacement Surgery of the Hip and Knee(2018-04-09) Damani, Zaheed; Marshall, Deborah A.; Noseworthy, Thomas Wm.; Quan, Hude; Bohm, Éric Richard; MacKean, Gail; Yelin, Edward H.; Hildebrand, Kevin A.Background: Single-entry is an increasingly prominent waiting time management strategy in healthcare but its impact on quality of care is poorly understood. We evaluated the Winnipeg Central Intake Service (WCIS) for total joint replacement (TJR) surgery of the hip or knee, a single-entry model (SEM) to manage patients referred for TJR surgery of the hip or knee. Methods: A pre/post mixed-methods case study approach was used to measure the WCIS' influence on six dimensions of quality of care: acceptability, accessibility, appropriateness, effectiveness, efficiency, safety. Qualitative interviews were used to assess experiences of patients, family physicians, orthopaedic surgeons, surgical office assistants and WCIS project team members. A pre/post intervention cross-sectional design was used to quantitatively assess changes in the six dimensions by comparing historical and prospective cohorts. Results: Our qualitative inquiry revealed that benefits of the WCIS included streamlined processes, greater patient access, improved measurement and monitoring of outcomes. Challenges included low awareness, change readiness, and initial participation among stakeholders. Unanticipated consequences included workload increases, confusion around stakeholder expectations, and under-reporting of data by surgeons' offices. Stakeholder acceptability was conditional, not universal. Assessment of capacity and readiness to change, and efforts to increase awareness, preparedness and uptake are critical. Factors for successful implementation include clear communication, robust data collection, physician leadership, and patience by all (especially implementation teams) allowing for an effective top-down, and bottom up approach. Our quantitative analysis revealed that the WCIS reduced variability across surgeon waiting times, with modest reductions in overall waits for surgery. There was improvement in some, but not all, dimensions of quality. Waiting time was significantly improved (WT) for consult for TJR of the hip (WT1) and all WTs for TJR of the knee. Total knee replacement surgeries performed within the nationally-recommended 26-week benchmark increased by 5.9% post-WCIS. Post-surgical complication rates (safety) were lower post-WCIS. Accessibility and safety were the only quality dimensions that changed (post-WCIS for TJR of the hip and knee). Conclusion: Overall, WCIS implementation contributed to improvement in some, but not all dimensions of quality of care. This is the first study to comprehensively assess the influence of SEMs on the delivery of TJRs across all dimensions of quality. Findings of this research are generally consistent with existing literature related to SEMs and change management in healthcare. SEMs show an ability to improve accessibility without adversely affecting other dimensions of quality, albeit with conditional, not universal stakeholder acceptability. Limitations of this study include non-longitudinal cohorts, and availability and quality of data. Findings from this research can help strengthen existing SEMs and inform development of new ones for improved patient experience and outcomes and system performance.Item Open Access A systematic approach to using regression modelling and ‘big data’ to derive a meaningful clinical decision rule for epilepsy(2018-08-22) Josephson, Colin Bruce; Wiebe, Samuel; Jetté, Nathalie; Sajobi, Tolulope T.; Marshall, Deborah A.Introduction: clinical decision rules (CDRs) have been developed in a number of medical fields resulting in improved patient outcomes, quality of care, and health economics. Aims: to identify all CDRs developed for epilepsy and to derive one that guides the prescription of the antiepileptic drug (AED), levetiracetam, according to its risk of a psychiatric adverse effect. Methods: a systematic review and meta-analysis was first performed to determine the state of the literature with respect to CDRs in epilepsy. The Health Improvement Network (THIN) electronic medical records register was used to identify patients with epilepsy by employing a modified validated case definition with a 5-year washout. Analyses were restricted to patients receiving AED monotherapy and the association between levetiracetam use and psychiatric adverse effects was explored Cox proportional hazards regression with timevarying covariates. Finally, logistic regression with parameter regularisation and k=5 fold cross validation was used to derive the CDR that predicts the development of psychiatric adverse effects following levetiracetam prescription. Results: the systematic review identified four epilepsy-specific CDRs, none of which guided AED prescription. A total of 9595 presumed incident cases of epilepsy (85.7 cases per 100,000 persons) were identified in THIN. Both carbamazepine (hazard ratio [HR]: 0.84, 95% confidence interval [95% CI]: 0.73– 0.97; p = 0.02) and lamotrigine (HR: 0.83, 95% CI: 0.70–0.99; p = 0.03) were associated with reduced hazards of a psychiatric sign, symptom, or disorder iii compared to no AED treatment. Levetiracetam was not associated with psychiatric adverse effects but the analyses were underpowered (n=202; 3%). All patients receiving levetiracetam (1173/7400; 16%) were included for CDR derivation. Prediction variables were incorporated into multiple logistic regression models with parameter regularisation. Odds of reporting a psychiatric complaint were elevated for females and those with a pre-exposure history of depression, anxiety, recreational drug use, or higher social deprivation. The prediction model performed well (area under the curve [AUC] 0.68; 95% confidence interval 0.58- 0.79 after stratified k=5 fold cross-validation). Using a cut-off threshold 0.1, the CDR had a specificity of 83%. Conclusion: If externally validated and properly implemented, this CDR could be used to guide prescription in clinical practice.Item Open Access A Systematic Review of the Cost-Effectiveness of Nurse Practitioners and Clinical Nurse Specialists: What Is the Quality of the Evidence?(2014-09-01) Donald, Faith; Kilpatrick, Kelley; Reid, Kim; Carter, Nancy; Martin-Misener, Ruth; Bryant-Lukosius, Denise; Harbman, Patricia; Kaasalainen, Sharon; Marshall, Deborah A.; Charbonneau-Smith, Renee; Donald, Erin E.; Lloyd, Monique; Wickson-Griffiths, Abigail; Yost, Jennifer; Baxter, Pamela; Sangster-Gormley, Esther; Hubley, Pamela; Laflamme, Célyne; Campbell–Yeo, Marsha; Price, Sheri; Boyko, Jennifer; DiCenso, AlbaBackground. Improved quality of care and control of healthcare costs are important factors influencing decisions to implement nurse practitioner (NP) and clinical nurse specialist (CNS) roles. Objective. To assess the quality of randomized controlled trials (RCTs) evaluating NP and CNS cost-effectiveness (defined broadly to also include studies measuring health resource utilization). Design. Systematic review of RCTs of NP and CNS cost-effectiveness reported between 1980 and July 2012. Results. 4,397 unique records were reviewed. We included 43 RCTs in six groupings, NP-outpatient (), NP-transition (), NP-inpatient (), CNS-outpatient (), CNS-transition (), and CNS-inpatient (). Internal validity was assessed using the Cochrane risk of bias tool; 18 (42%) studies were at low, 17 (39%) were at moderate, and eight (19%) at high risk of bias. Few studies included detailed descriptions of the education, experience, or role of the NPs or CNSs, affecting external validity. Conclusions. We identified 43 RCTs evaluating the cost-effectiveness of NPs and CNSs using criteria that meet current definitions of the roles. Almost half the RCTs were at low risk of bias. Incomplete reporting of study methods and lack of details about NP or CNS education, experience, and role create challenges in consolidating the evidence of the cost-effectiveness of these roles.Item Open Access Are we Capturing the Socioeconomic Burden of Rare Genetic Disease? A Scoping Review of Economic Evaluation and Cost-of-Illness Studies(PharmacoEconomics, 2023-12) Marshall, Deborah A.; Gerber, Brittany; Lorenzetti, Diane L.; MacDonald, Karen V.; Bohach, Riley J.; Currie, Gillian R.Objectives: Rare diseases have a significant impact on patients, families, the health system, and society. Measuring the socioeconomic burden (SEB) is crucial to valuing interventions for rare diseases. Healthcare system costs are significant, but so are costs to other government sectors, patients, families, and society. To understand the breadth of costs captured in rare disease studies, we examined the cost categories and elements of SEB captured in published studies. Methods: A scoping review was conducted using five electronic databases to identify English language economic evaluations and cost-of-illness studies of interventions for rare diseases (2011-2021). We mapped costs using a previously developed evidence-informed framework of SEB costs for rare disease. Results: Of 4890 studies identified, 48 economic evaluations and 22 cost-of-illness studies were included. While 18/22 cost-of-illness studies utilized a societal perspective, only 7/48 economic evaluations incorporated societal costs. Most reported cost categories related to medical costs, with medication and hospitalizations being the most common elements for both study designs. Costs borne by patients, families, and society were reported less among economic evaluations than cost-of-illness studies. These included: productivity (10% vs 77%), travel/accommodation (6% vs 68%), government benefits (4% vs 18%), and family impacts (0% vs 50%). Conclusions: Contrary to cost-of-illness analyses, most of the included economic evaluations did not account for the hidden burden of rare disease, that is costs borne by patients, families, and societies. Including these types of costs in future studies would provide a more comprehensive picture of the burden of disease, providing empirical data to inform how we value and make decisions regarding rare disease interventions, health policy and resource allocation.Item Open Access Assessing Health Care Costs, Treatment Patterns, and Health-Related Quality of Life in Children with Juvenile Idiopathic Arthritis and their Caregivers.(2022-01-20) Grazziotin Lago, Luiza R.; Marshall, Deborah A.; Twilt, Marinka; Currie, Gillian; Koffijberg, HendrikJuvenile idiopathic arthritis (JIA) is an umbrella term that encompasses all forms of arthritis with onset before the age of 16 years and symptoms that persist for more than 6 weeks for which the cause is unknown. JIA can significantly affect the quality of life of children and their families, and the disease and its treatment can increase the economic burden for the health care system and families.This PhD thesis was focused on a research program that addressed four gaps in the literature: 1) assessment of the overall and JIA-related health care resource utilization and associated costs before and after rheumatology care; 2) evaluation of JIA treatment prescription patterns; 3) studies measuring health state utility scores of patients with JIA across their lifespan, including children, adults, and adolescents; and 4) measurement of caregiver health-related quality of life (HRQoL) and care-related quality of life (CRQoL) and investigation of the relationship of those measures with child HRQoL and other influencing factors.We conducted four studies to address those gaps. Two retrospective cohort studies using electronic medical records and administrative databases to assess health care resource use and costs, and sequences of treatment prescriptions in children with JIA in Canada, respectively. Our findings reveal that the care pathway for children with JIA including health care resource utilization, costs, and treatment prescription patterns can be expensive, and complex – and varies by JIA subtype. We then performed a systematic review, which was the first review seeking to identify and assess health utility scores in children, adolescents, and adults with history of JIA. Finally, our last study was the first to assess care-related quality of life in caregivers of children with JIA, and to evaluate factors associated with caregiver care- and health-related quality of life, which highlighted the need for an encompassing family-centred approach of care that goes beyond achieving inactive disease.Item Open Access Barriers experienced by families new to Alberta, Canada when accessing routine-childhood vaccinations(2023-07-12) Fullerton, Madison M.; Pateman, Margaret; Hasan, Hinna; Doucette, Emily J.; Cantarutti, Stephen; Koyama, Amanda; Weightman, Amanda M.; Tang, Theresa; Coakley, Annalee; Currie, Gillian R.; Fabreau, Gabriel; Constantinescu, Cora; Marshall, Deborah A.; Hu, JiaAbstract Background As Canada and other high-income countries continue to welcome newcomers, we aimed to 1) understand newcomer parents’ attitudes towards routine-childhood vaccinations (RCVs), and 2) identify barriers newcomer parents face when accessing RCVs in Alberta, Canada. Methods Between July 6th—August 31st, 2022, we recruited participants from Alberta, Canada to participate in moderated focus group discussions. Inclusion criteria included parents who had lived in Canada for < 5 years with children < 18 years old. Focus groups were transcribed verbatim and analyzed using content and deductive thematic analysis. The capability opportunity motivation behaviour model was used as our conceptual framework. Results Four virtual and three in-person focus groups were conducted with 47 participants. Overall, parents were motivated and willing to vaccinate their children but experienced several barriers related to their capability and opportunity to access RCVs. Five main themes emerged: 1) lack of reputable information about RCVs, 2) language barriers when looking for information and asking questions about RCVs, 3) lack of access to a primary care provider (PCP), 4) lack of affordable and convenient transportation options, and 5) due to the COVID-19 pandemic, lack of available vaccine appointments. Several minor themes were also identified and included barriers such as lack of 1) childcare, vaccine record sharing, PCP follow-up. Conclusions Our findings highlight that several barriers faced by newcomer families ultimately stem from issues related to accessing information about RCVs and the challenges families face once at vaccination clinics, highlighting opportunities for health systems to better support newcomers in accessing RCVs.Item Open Access Developing a Framework of Cost Elements of Socioeconomic Burden of Rare Disease: A Scoping Review(PharmacoEconomics, 2023-04-07) Currie, Gillian R; Gerber, Brittany; Lorenzetti, Diane; MacDonald, Karen; Benseler, Susanne M; Bernier, Francois P; Boycott, Kym M; Carias, K. Vanessa; Hamelin, Bettina; Hayeems, Robin Z; LeBlanc, Claire; Twilt, Marinka; van Rooijen, Gijs; Wong-Rieger, Durhane; Yeung, Rae S. M.; Marshall, Deborah A.Background and Objective: Rare diseases place a significant burden on patients, families, the healthcare system and society. Evidence on the socioeconomic burden of rare disease is limited and mostly reflects diseases where treatments are available. We developed a framework encompassing recommended cost elements for studies of the socioeconomic burden of rare diseases. Methods: A scoping review, conducted in five databases (Cochrane Library, EconLit, Embase, MEDLINE, and APA PsycINFO), identified English language publications from 2000 and 2021 presenting frameworks developed for determining, measuring or valuing costs for rare or chronic diseases. Cost elements were extracted and used to develop a literature-informed framework. Structured feedback was gathered from experts in rare diseases, health economics/health services and policy research to revise the framework. Results: Of 2990 records identified, eight papers were included and informed our preliminary framework; three focused on rare disease and five on chronic disease. Following expert input, we developed a framework consisting of nine cost categories (inpatient, outpatient, community, healthcare products/goods, productivity/education, travel/accommodation, government benefits, family impacts, and other), with several cost elements within each category. Our framework includes unique costs, added from the expert feedback, including genetic testing to inform treatment, use of private laboratories or out-of-country testing, family involvement in foundations and organizations, and advocacy costs for special access programs. Conclusions: Our work is the first to identify a comprehensive list of cost elements for rare disease for use by researchers and policy makers to fully capture socioeconomic burden. Use of the framework will increase the quality and comparability of future studies. Future work should focus on measuring and valuing these costs through onset, diagnosis, and post-diagnosis.Item Open Access An Economic Evaluation of Body Checking Policies in Bantam Ice Hockey(2019-01-25) Lee, Raymond; Currie, Gillian R.; Marshall, Deborah A.; Emery, Carolyn A.; Palacios-Derflingher, Luz MariaSport-related injury is the leading cause of injury in youth and are costly to the healthcare system. Disallowing body checking in Pee Wee (ages 11-12) ice hockey has been found to be effective in reducing the risk of injuries and associated healthcare costs, however the impact on injury risk and costs in Bantam (ages 13-14) remains unknown. The objectives of this study are to compare injury rates and costs between non-elite (lower 70% divisions of play) Bantam players in leagues allowing body checking to where body checking is disallowed, and to project the overall change on the number of injuries and costs to the Alberta healthcare system if body checking were disallowed for all Bantam players over one season. The study found that disallowing body checking reduced injuries by 4.32/1000 player-hours and saved cost by $1,737/1000 player-hours in the public healthcare system. This policy change could potentially prevent 1,102 injuries that occur during games and save $331,522 in the public healthcare system over one season in Alberta. However, this study used injury rates adjusted only for exposure hours and team clustering, but not other covariates or repeated observations. Thus further analysis is required before policy recommendations can be made.Item Open Access Evaluating key performance indicators of the process of care in juvenile idiopathic arthritis(2023-04-21) Cooper, Sarah M.; Currie, Gillian R.; Kromm, Seija; Twilt, Marinka; Marshall, Deborah A.Abstract Objective To determine whether and how often the information to measure a set of key performance indicators (KPIs) in juvenile idiopathic arthritis (JIA) is found in data collected routinely in a Pediatric Rheumatology Clinic. Methods A retrospective electronic chart review and administrative data analysis was conducted for a cohort of 140 patients with JIA at a tertiary Pediatric Rheumatology Clinic between 2016–2020. The set of KPIs include measuring patient outcomes (joint assessment, physician’s global assessment of disease activity, assessment of functional ability, composite disease activity measurement), access to care (waiting time between referral and first visit, visit with the rheumatologist within the first year of diagnosis, annual follow-up visits with the rheumatologist), and safety (tuberculosis screening, and laboratory monitoring). Documentation was assessed as a binary variable indicating whether the required information was ever found. Documentation frequency for each KPI was assessed with counts and percentages of the number of times the required information was documented for each clinic visit. Compliance with the safety KPI definitions was assessed using administrative databases. Results Data for each KPI were found at least once in the cohort and documentation varied in frequency and consistency. Access to care and safety KPIs were documented more frequently than patient outcome KPIs. A joint assessment was documented at every visit for 95% of patients, 46% for an assessment of pain, and none for a physician’s global assessment of disease activity, an assessment of functional ability, or a composite disease activity measurement. Conclusion Although feasible to measure, there is an opportunity for improving the consistency of documentation. Having an active system of monitoring KPIs and tools to simplify measurement is a key step in the process toward improved patient care outcomes. Streamlining the collection of KPI data can increase the likelihood of compliance. Next steps should involve replicating this study in various centres.Item Open Access Exploring Health Locus of Control and Patient Preferences for the Non-Surgical Management of Osteoarthritis(2021-08-09) Kennedy, Bryanne L; Currie, Gillian R.; Marshall, Deborah A.; Emery, Carolyn A.; Kania-Richmond, Anna A.; Gagliardi, Anna R.Background:There is no cure for osteoarthritis, and management aims to reduce pain and improve function. Despite guidelines, 61% of people are referred for surgery before trying non-surgical options, and this needs to be further understood. Many factors affect uptake of options, including the osteoarthritis information available to patients. Individual beliefs around who and whatinfluences health, or locus of control, is related to patient preferences for disease management. There is a paucity of literature on locus of control and preferences for osteoarthritis management.Objectives:To identify the factors that patients with hip and knee osteoarthritis consider when choosing management strategies, and to explore their beliefs about who and what influences osteoarthritis symptoms and progression.Methods:Semi-structured interviews with patients who self-reported a diagnosis by a medical professional with hip and/or knee osteoarthritis and had at least one osteoarthritic joint that had not undergone replacement. A multifaceted recruitment approach was used. Interviews were transcribed verbatim and the Braun and Clarke method for thematic analysis was employed.Results:Interviews were conducted with 13 patients from Alberta, Canada. Sixteen factors were identified that participants considered when choosing management strategies. Majority were extrinsic (11), relating to features of programs and services, while five were intrinsic relating to how individual perspectives influence decision making. Three novel factors emerged: 1) whether further management was desired, 2) the prospect of surgery, and 3) participants’ choice inmanagement. Three descriptive themes captured participants’ beliefs about who and what influences their osteoarthritis symptoms and progression: etiology, progression, and symptoms. Most participants believed that others and themselves had some influence over their osteoarthritis symptoms.Conclusions:The findings reinforced that patient’s management decisions for their osteoarthritis were based on the options available to them. This was the first study to explore locus of control in the context of non-surgical osteoarthritis management and provides a point of departure for future work. Factors that patients consider when choosing management strategies were also identifiedthat could be considered for subsequent quantitative research on patient preferences.Keywords: osteoarthritis, internal-external control, patient preference, conservative treatment,disease management, qualitative researchItem Open Access Exploring patient perspectives on EQ-5D-5L data visualization within an individualized decision aid for total knee arthroplasty (TKA) in Alberta, Canada(2024-02-29) Johnson, Jeffrey A.; Itiola, Ademola; Rahman, Shakib; Smith, Christopher; Soprovich, Allison; Wozniak, Lisa A.; Marshall, Deborah A.Abstract Background Decision aids can help patients set realistic expectations. In this study, we explored alternative presentations to visualise patient-reported outcomes (EQ-5D-5L) data within an online, individualized patient decision aid for total knee arthroplasty (TKA) that, in part, generates individualized comparisons based on age, sex and body mass index, to enhance usability prior to implementation into routine clinical practice. Methods We used data visualization techniques to modify the presentation of EQ-5D-5L outcomes data within the decision aid. The EQ-5D-5L data was divided into two parts allowing patients to compare themselves to similar individuals (1) pre-surgery and (2) 1-year post-surgery. We created 2 versions for each part and sought patient feedback on comprehension, usefulness, and visual appeal. Patients from an urban orthopedic clinic were recruited and their ratings and comments were recorded using a researcher-administered checklist. Data were managed using Microsoft Excel, R version 3.6.1 and ATLAS.ti V8 and analyzed using descriptive statistics and directed content analysis. Results A total of 24 and 25 patients participated in Parts 1 and 2, respectively. Overall, there was a slight preference for Version 1 in Part 1 (58.3%) and Version 2 in Part 2 (64%). Most participants demonstrated adequate comprehension for all versions (range 50–72%) and commented that the instructions were clear. While 50–60% of participants rated the content as useful, including knowing the possible outcomes of surgery, some participants found the information interesting only, were unsure how to use the information, or did not find it useful because they had already decided on a treatment. Participants rated visual appeal for all versions favorably but suggested improvements for readability, mainly larger font and image sizes and enhanced contrast between elements. Conclusions Based on the results, we will produce an enhanced presentation of EQ-5D-5L data within the decision aid. These improvements, along with further usability testing of the entire decision aid, will be made before implementation of the decision aid in routine clinical practice. Our results on patients’ perspectives on the presentation of EQ-5D-5L data to support decision making for TKA treatments contributes to the knowledge on EQ-5D-5L applications within healthcare systems for clinical care.Item Open Access Factors associated with care- and health-related quality of life of caregivers of children with juvenile idiopathic arthritis(2022-07-23) Grazziotin, Luiza R.; Currie, Gillian; Twilt, Marinka; IJzerman, Maarten J.; Kip, Michelle M. A.; Koffijberg, Hendrik; Bonsel, Gouke; Benseler, Susanne M.; Swart, Joost F.; Vastert, Sebastiaan J.; Wulffraat, Nico M.; Yeung, Rae S. M.; Armbrust, Wineke; van den Berg, J. M.; Marshall, Deborah A.Abstract Objective This study investigates the relationship of child, caregiver, and caring context measurements with the care-related quality of life (CRQoL) and health-related quality of life (HRQoL) of caregivers of children with juvenile idiopathic arthritis (JIA). Methods We performed a cross-sectional analysis of baseline data on caregivers of children with JIA from Canada and the Netherlands collected for the “Canada-Netherlands Personalized Medicine Network in Childhood Arthritis and Rheumatic Diseases” study from June 2019 to September 2021. We used the CRQoL questionnaire (CarerQoL), adult EQ-5D-5L, and proxy-reported Youth 5-Level version of EuroQoL (EQ-5D-5L-Y) to assess caregiver CRQoL, caregiver HRQoL, and child HRQoL, respectively. We used a multivariate analysis to assess the relationship between both caregiver CRQoL and HRQoL and patient, caregiver, and caring context measurements. Results A total of 250 caregivers were included in this study. Most of the caregivers were from the Netherlands (n = 178, 71%) and 77% were females (n = 193). The mean CarerQoL scores was 82.7 (standard deviation (SD) 11.4) and the mean EQ-5D-5L utility score was 0.87 (SD 0.16). Child HRQoL and employment had a positive relationship with both caregiver CarerQoL and EQ-5D-5L utility scores (p < 0.05), while receiving paid or unpaid help had a negative relationship with both scores (p < 0.05). Conclusion Our findings indicated that to understand the impact of JIA on families, we need to consider socio-economic factors, such as employment and support to carry caregiving tasks, in addition to child HRQoL.Item Open Access Key ingredients for successful collaboration in health research: perspectives of patient research partners(2024-06-05) Bruce, Marcia; Yogaratnam, Karthika; Suryaprakash, Nitya; Barker, Karis L.; Marshall, Deborah A.Abstract Background There are increasing publications on meaningful collaboration between researchers and patient research partners (PRPs), but fewer publications of such work from the PRP perspective using an evaluation framework. Our aim is to present our own perspectives and reflections on meaningful collaboration as PRPs working on a qualitative research study. Main body We were part of a study team that comprised of PRPs, clinicians and academic researchers, and was led by a PRP. The team designed and conducted a qualitative study aimed at understanding how patients make decisions around tapering of biologics for inflammatory bowel disease. The study was conducted online. The PRP lead was trained in qualitative methodology through a one-year certificate program called Patient and Community Engagement Research offered through the University of Calgary Continuing Education. We had received patient-oriented research training and qualitative research training prior to this project. Team members were assigned tasks by our group lead based on member interests and willingness. Some group members were part of the Strategy for Patient-Oriented Research, Inflammation, Microbiome, and Alimentation: Gastro-Intestinal and Neuropsychiatric Effects Network, one of five chronic disease networks in the Strategy for Patient Oriented Research initiative of the Canadian Institutes of Health Research. We describe the five key ingredients to successful collaboration based on our experiences and reflections utilizing the Experience-Reflection-Action Cycle as our framework. The five key ingredients that we identified were: inclusiveness, goal and role clarity, multi-level training and capacity building, shared decision making, and a supportive team lead. Conclusion Overall, our experience was positive. With successful collaboration came an increased level of trust, commitment and performance. There is a need for more studies with diverse PRPs in different settings to validate and/or identify additional factors to improve collaboration in patient-oriented research.Item Open Access Queueing analysis of two healthcare systems where physician activity is offloaded to supporting health professionals(2020-06-30) Tagimacruz, Maria Antonieta; Bischak, Diane P.; Marshall, Deborah A.; Bijvank, Marco; Brennan, Robert William; da Silveira, Giovani J. C.; Willoughby, K. A.Patient waiting time to see a specialist and specialist utilization are vital aspects of providing musculoskeletal (MSK) healthcare service. Long waiting times not only subject patients to physical pain and suffering but also detrimentally impacts society. This dissertation uses queuing theory to analyze three models for rheumatologists consultation involving an alternative care provider, osteoarthritis consultation involving a musculoskeletal screener, and a central intake referral system for osteoarthritis patients. The second chapter presents a queueing and simulation approach to analyze the impact of an alternative care provider in a rheumatologist consultation system on waiting time and specialist utilization. Using a multi-class closed queueing network, we look into the boundaries for workload allocation within which performance improvements are realized. Chapter three compares an osteoarthritis consultation system with and without an MSK screener modeled as a network of queues. In addition to the impact of the addition of the MSK screener, we also investigate the impact of the surgical threshold of the screener relative to that of the surgeon to the patient waiting time and surgeon’s utilization. Finally, in the fourth chapter, we model an osteoarthritis referral central intake system and explore the impact of the deterministic and probabilistic routing decisions at the central intake on surgical patient waiting time and surgeon’s utilization.Item Open Access Real-world data reveals the complexity of disease modifying anti-rheumatic drug treatment patterns in juvenile idiopathic arthritis: an observational study(2022-04-11) Grazziotin, Luiza R.; Currie, Gillian; Twilt, Marinka; Ijzerman, Maarten J.; Kip, Michelle M. A.; Koffijberg, Hendrik; Benseler, Susanne M.; Swart, Joost F.; Vastert, Sebastiaan J.; Wulffraat, Nico M.; Yeung, Rae S. M.; Marshall, Deborah A.Abstract Objective Pharmacological treatment is a cornerstone of care for children with juvenile idiopathic arthritis (JIA). The objective of this study is to evaluate prescription patterns of conventional and biologic disease modifying anti-rheumatic drugs (c-DMARDs and b-DMARDs) for patients with JIA. Methods We conducted a retrospective cohort study of children diagnosed with JIA at a rheumatology pediatric clinic. Eligibility criteria were defined as children and youth newly diagnosed with enthesis-related arthritis, polyarticular, or oligoarticular JIA between 2011 and 2019, with at least one year of observation. Data on c-DMARDs and b-DMARDs prescriptions were obtained from electronic medical charts. We used descriptive statistics, Kaplan–Meier survival methods, and Sankey diagrams to describe treatment prescription patterns. Results A total of 325 patients with JIA were included, with a median observation time of 3.7 years. The most frequently prescribed c-DMARD and b-DMARD were methotrexate and etanercept, respectively. Within the first year of rheumatology care, 62% and 21% of patients had a c-DMARD and a b-DMARD prescribed, respectively. These proportions varied greatly by JIA subtype. Among the 147 (147/325, 45%) patients that had at least one b-DMARD prescribed, 24% were prescribed a second, and 7% a third-line of b-DMARD. A total of 112 unique treatment sequences were observed, with c-DMARD monotherapy followed by the addition of either a b-DMARD (56%) or another c-DMARD (30%) being the two most prevalent patterns in this cohort. Conclusion We observed a variety of treatment trajectories, with many patients experiencing multiple treatment lines, illustrating the complexity of the overall JIA treatment path.Item Open Access System-level performance measures of access to rheumatology care: a population-based retrospective study of trends over time and the impact of regional rheumatologist supply in Ontario, Canada, 2002–2019(2022-12-27) Barber, Claire E.; Lacaille, Diane; Croxford, Ruth; Barnabe, Cheryl; Marshall, Deborah A.; Abrahamowicz, Michal; Xie, Hui; Avina-Zubieta, J. A.; Esdaile, John M.; Hazlewood, Glen; Faris, Peter; Katz, Steven; MacMullan, Paul; Mosher, Dianne; Widdifield, JessicaAbstract Objective To determine whether there were improvements in rheumatology care for rheumatoid arthritis (RA) between 2002 and 2019 in Ontario, Canada, and to evaluate the impact of rheumatologist regional supply on access. Methods We conducted a population-based retrospective study of all individuals diagnosed with RA between January 1, 2002 and December 31, 2019. Performance measures evaluated were: (i) percentage of RA patients seen by a rheumatologist within one year of diagnosis; and (ii) percentage of individuals with RA aged 66 years and older (whose prescription drugs are publicly funded) dispensed a disease modifying anti-rheumatic drug (DMARD) within 30 days after initial rheumatologist visit. Logistic regression was used to assess whether performance improved over time and whether the improvements differed by rheumatology supply, dichotomized as < 1 rheumatologist per 75,000 adults versus ≥1 per 75,000. Results Among 112,494 incident RA patients, 84% saw a rheumatologist within one year: The percentage increased over time (adjusted odds ratio (OR) 2019 vs. 2002 = 1.43, p < 0.0001) and was consistently higher in regions with higher rheumatologist supply (OR = 1.73, 95% CI 1.67–1.80). Among seniors who were seen by a rheumatologist within 1 year of their diagnosis the likelihood of timely DMARD treatment was lower among individuals residing in regions with higher rheumatologist supply (OR = 0.90 95% CI 0.83–0.97). These trends persisted after adjusting for other covariates. Conclusion While access to rheumatologists and treatment improved over time, shortcomings remain, particularly for DMARD use. Patients residing in regions with higher rheumatology supply were more likely to access care but less likely to receive timely treatment.Item Open Access The use of patient-reported outcome measures in hip and knee arthroplasty in Alberta(2021-10-12) Marshall, Deborah A.; Jin, Xuejing; Pittman, Lindsay B.; Smith, Christopher J.Abstract PROMs are part of routine measurement for hip and knee replacement in Alberta, Canada. We provide an overview of how PROMs are implemented in routine care, and how we use PROMs data for decision-making at different levels within the health system. The Alberta Bone and Joint Health Institute (ABJHI) ran a randomized controlled trial to determine the effectiveness and cost-effectiveness of an evidence-based care pathway for hip and knee arthroplasty in 2004. The study included several PROMs questionnaires: Western Ontario and McMaster Universities Osteoarthritis Index, Health Utility Index, Short Form 36 and the EQ-5D-3L. Subsequently, the focus shifted to spread and scale of the care pathway provincially. WOMAC and EQ-5D-3L and a patient experience survey were selected for provincial adoption – captured before surgery, three-months post-surgery, and 12-months post-surgery. These PROMs data were integrated into research and routine clinical practice at the micro, meso and macro levels. At the micro level, PROMs data are used at the individual patient and provider level for patients to provide input on their care and as a tool to communicate with their healthcare providers. We examined the relationship of appropriateness and patient reported outcomes in a prospective cohort study. We evaluated whether routinely collected PROMs could be integrated into a patient decision aid to better inform shared decision making. At the meso level, continuous quality improvement reports are provided routinely to individual health care providers, hospitals and clinics on their performance against the measurement framework and standard key performance indicators. At the macro level, PROMs data are used to evaluate system performance by comparing outcomes across different jurisdictions or over time and support health policy decision making. Combined with administrative databases, we have used simulation models to reflect transition through the continuum of care from disease onset through end-stage care regarding the burden of disease, healthcare resource requirements and associated healthcare costs. The addition of PROMs data in clinical repositories and analyses enables the system to identify and address issues of continuous quality improvement against a measurement framework of performance indicators and to explicitly recognize the trade-offs that are inherent in any resource-constrained system.Item Open Access Uncovering the hidden socioeconomic impact of juvenile idiopathic arthritis and paving the way for other rare childhood diseases: an international, cross-disciplinary, patient-centered approach (PAVE Consortium)(2024-08-09) Marshall, Deborah A.; Gerber, Brittany; Currie, Gillian R.; Antón, Jordi; De Somer, Lien; Dey, Michelle; Egert, Tsipi; Egert, Yona; Henan, Lia; Klotsche, Jens; Mifsut, Laura M.; Minden, Kirsten; Normand, Christophe; Porte, David; Saurenmann, Rotraud K.; Swart, Joost F.; Uziel, Yosef; Wilson, Jennifer; Wouters, Carine; Ziv, Amit; Benseler, Susanne M.Abstract Background Juvenile idiopathic arthritis (JIA) refers to a heterogeneous group of rheumatic conditions in children. Novel drugs have greatly improved disease outcomes; however, outcomes are impacted by limited awareness of the importance of early diagnosis and adequate treatment, and by differences in access across health systems. As a result, patients with JIA continue to be at risk for short- and long-term morbidity, as well as impacts on virtually all aspects of life of the child and family. Main body Literature on the socioeconomic burden of JIA is largely focused on healthcare costs, and the impact of JIA on patients, families, and communities is not well understood. High quality evidence on the impact of JIA is needed to ensure that patients are receiving necessary support, timely diagnostics, and adequate treatment, and to inform decision making and resource allocation. This commentary introduces the European Joint Programme on Rare Diseases: Producing an Arthritis Value Framework with Economic Evidence: Paving the Way for Rare Childhood Diseases (PAVE) project, which will co-develop a patient-informed value framework to measure the impact of JIA on individuals and on society. With a patient-centered approach, fundamental to PAVE is the involvement of three patient advocacy organizations from Canada, Israel, and Europe, as active research partners co-designing all project phases and ensuring robust patient and family engagement. The framework will build on the findings of projects from six countries: Canada, Germany, Switzerland, Spain, Israel, and Belgium, exploring costs, outcomes (health, well-being), and unmet needs (uveitis, mental health, equity). Conclusion This unique international collaboration will combine evidence on costs (from family to societal), outcomes (clinical, patient and family outcomes), and unmet needs, to co-design and build a framework with patients and families to capture the full impact of JIA. The framework will support the development of high-quality evidence, encompassing economic and clinical considerations, unmet needs, and patient perspectives, to inform equitable resource allocation, health system planning, and quality of care better aligned with the needs of children with JIA, their families, and communities. Knowledge gained from this novel approach may pave the way forward to be applied more broadly to other rare childhood diseases.Item Open Access Withdrawing biologics in non-systemic JIA: what matters to pediatric rheumatologists?(2023-07-11) van Til, Janine A.; Kip, Michelle M. A.; Schatorjé, Ellen J. H.; Currie, Gillian; Twilt, Marinka; Benseler, Susanne M.; Swart, Joost F.; Vastert, Sebastiaan J.; Wulffraat, Nico; Yeung, Rae S. M.; Groothuis-Oudshoorn, C. G. M. (.; Warta, Sanne; Marshall, Deborah A.; IJzerman, Maarten J.Abstract Objective Approximately one third of children with JIA receive biologic therapy, but evidence on biologic therapy withdrawal is lacking. This study aims to increase our understanding of whether and when pediatric rheumatologists postpone a decision to withdraw biologic therapy in children with clinically inactive non-systemic JIA. Methods A survey containing questions about background characteristics, treatment patterns, minimum treatment time with biologic therapy, and 16 different patient vignettes, was distributed among 83 pediatric rheumatologists in Canada and the Netherlands. For each vignette, respondents were asked whether they would withdraw biologic therapy at their minimum treatment time, and if not, how long they would continue biologic therapy. Statistical analysis included descriptive statistics, logistic and interval regression analysis. Results Thirty-three pediatric rheumatologists completed the survey (40% response rate). Pediatric rheumatologists are most likely to postpone the decision to withdraw biologic therapy when the child and/or parents express a preference for continuation (OR 6.3; p < 0.001), in case of a flare in the current treatment period (OR 3.9; p = 0.001), and in case of uveitis in the current treatment period (OR 3.9; p < 0.001). On average, biologic therapy withdrawal is initiated 6.7 months later when the child or parent prefer to continue treatment. Conclusion Patient’s and parents' preferences were the strongest driver of a decision to postpone biologic therapy withdrawal in children with clinically inactive non-systemic JIA and prolongs treatment duration. These findings highlight the potential benefit of a tool to support pediatric rheumatologists, patients and parents in decision making, and can help inform its design.