(Un)standardized testing: the diagnostic odyssey of children with rare genetic disorders in Alberta, Canada.

PURPOSE: We provide a description of the diagnostic odyssey for a cohort of children seeking diagnosis of a rare genetic disorder in terms of the time from initial consultation to most recent visit or receipt of diagnosis, the number of tests per patient, and the types of tests received. METHODS: Retrospective chart review of 299 children seen at the Alberta Children's Hospital (ACH) Genetics Clinic (GC) for whom the result of at least one single-gene test, gene panel, or chromosome microarray analysis (CMA) was recorded. RESULTS: Of 299 patients, 90 (30%) received a diagnosis in the period of the review. Patients had an average of 5.4 tests each; 236 (79%) patients received CMA; 172 (58%) patients received single-gene tests and 34 (11%) received gene panels; 167 (56%) underwent imaging/electrical activity studies. The mean observation period was 898 days (95% confidence interval [CI] 791, 1004). Among patients with visits recorded prior to visiting ACH GC, 43% of the total observation time occurred prior to the GC. CONCLUSION: As genomic technologies expand, the nature of the diagnostic odyssey will change. This study has outlined the current standard of care in the ACH GC, providing a baseline against which future changes can be assessed.

Correction: The value of diagnostic testing for parents of children with rare genetic diseases.

An amendment to this paper has been published and can be accessed via a link at the top of the paper.

Direct health-care costs for children diagnosed with genetic diseases are significantly higher than for children with other chronic diseases.

PURPOSE: We aimed to estimate direct health-care costs and physician utilization for a cohort of children diagnosed with genetic diseases. METHODS: Retrospective cohort study using population-based provincial health administrative data for children with genetic diseases (n = 255) compared with three matched cohorts (asthma n = 1275, diabetes n = 255, general population n = 1275). We estimated direct health-care costs and resource use 5 years after diagnosis in five categories: physician billing, same day surgery, emergency, inpatient hospitalizations, and home care. RESULTS: During the postdiagnostic period, annual mean total costs for the genetic disease cohort were significantly higher than all other cohorts. Annual mean total costs for all cohorts were highest in the year after diagnosis with costs for the genetic disease cohort between 4.54 and 19.76 times higher during the 5 years. Inpatient hospitalizations and physician billing accounted for the majority of costs. The genetic disease cohort received more care from specialists, whereas the chronic disease cohorts received more care from general practitioners. CONCLUSION: Direct health-care costs for children with genetic diseases are significantly higher than children with/without a chronic disease, particularly in the year after diagnosis. These findings are important when considering resource allocation and funding prioritization for children with genetic diseases.

The value of diagnostic testing for parents of children with rare genetic diseases.

PURPOSE: Exome sequencing (ES) can rapidly identify disease-causing variants responsible for rare, single-gene diseases, and potentially reduce the duration of the diagnostic odyssey. Our study examines how parents and families value ES. METHODS: We developed a discrete choice experiment (DCE) survey that was administered to parents of children with rare diseases. The DCE included 14 choice tasks with 6 attributes and 3 alternatives. A valuation-space model was used to estimate willingness to pay, willingness to wait for test results, and minimum acceptable chance of a diagnosis for changes in each attribute. RESULTS: There were n = 319 respondents of whom 89% reported their child had genetic testing, and 66% reported their child had a diagnosis. Twenty-six percent reported that their child had been offered ES. Parents were willing to pay CAD$6590 (US$4943), wait 5.2 years to obtain diagnostic test results, and accept a reduction of 3.1% in the chance of a diagnosis for ES compared with operative procedures. CONCLUSION: Timely access to ES could reduce the diagnostic odyssey and associated costs. Before ES is incorporated routinely into care for patients with rare diseases in Canada and more broadly, there must be a clear understanding of its value to patients and families.

An individualized patient-reported outcome measure (PROM) based patient decision aid and surgeon report for patients considering total knee arthroplasty: protocol for a pragmatic randomized controlled trial.

BACKGROUND: While the rates of total knee arthroplasty (TKA) continue to rise worldwide, there are concerns about whether all surgeries are appropriate. Guidelines for appropriateness suggest that patients should have realistic expectations for total knee arthroplasty (TKA), and that the patient and their surgeon should agree that the potential benefits outweigh the potential harms. The objective of this study is to evaluate whether routinely collected pre- and post-TKA patient-reported outcome measures (PROMs) could be integrated into a patient decision aid to better inform these appropriateness criteria. This randomised trial will evaluate the preliminary efficacy of a tailored PROM-based patient decision aid and surgeon report (compared to usual care) for patients considering TKA on decision quality. METHODS: This is a pragmatic, randomised controlled trial conducted at one site in Alberta, Canada. Adults over the age of 30 years, who have been scheduled for a TKA consultation at the Edmonton Bone and Joint Centre with a participating surgeon, who understand, speak, and read English, and can provide informed consent, are eligible to participate. Participants will be randomised to receive a PROM-based patient decision aid and surgeon report before their surgical consultation or usual care. The decision aid will provide patients with information on their expected outcomes based on the EQ-5D-5L PROM, and these estimates are individualized based on clinical and demographic characteristics. The primary outcome of this trial is decision quality. Analysis will consider outcomes intention to treat, and feasibility outcomes for implementing the trial to routine practise. DISCUSSION: This patient decision aid and surgeon report intervention could contribute to improved treatment decision-making for patients considering total knee arthroplasty. TRIAL REGISTRATION (REGISTRY AND NUMBER): ClinicalTrials.gov : NCT03240913. Registered on August 1, 2017.

Corrigendum: What are people willing to pay for whole-genome sequencing information, and who decides what they receive?

This corrects the article DOI: 10.1038/gim.2016.61.

Estimating Preferences for Complex Health Technologies: Lessons Learned and Implications for Personalized Medicine.

We examine key study design challenges of using stated-preference methods to estimate the value of whole-genome sequencing (WGS) as a specific example of genomic testing. Assessing the value of WGS is complex because WGS provides multiple findings, some of which can be incidental in nature and unrelated to the specific health concerns that motivated the test. In addition, WGS results can include actionable findings (variants considered to be clinically useful and can be acted on), findings for which evidence for best clinical action is not available (variants considered clinically valid but do not meet as high of a standard for clinical usefulness), and findings of unknown significance. We consider three key challenges encountered in designing our national study on the value of WGS-layers of uncertainty, potential downstream consequences with endogenous aspects, and both positive and negative utility associated with testing information-and potential solutions as strategies to address these challenges. We conceptualized the decision to acquire WGS information as a series of sequential choices that are resolved separately. To determine the value of WGS information at the initial decision to undergo WGS, we used contingent valuation questions, and to elicit respondent preferences for reducing risks of health problems and the consequences of taking the steps to reduce these risks, we used a discrete-choice experiment. We conclude by considering the implications for evaluating the value of other complex health technologies that involve multiple forms of uncertainty.

What are people willing to pay for whole-genome sequencing information, and who decides what they receive?

PURPOSE: Whole-genome sequencing (WGS) can be used as a powerful diagnostic tool as well as for screening, but it may lead to anxiety, unnecessary testing, and overtreatment. Current guidelines suggest reporting clinically actionable secondary findings when diagnostic testing is performed. We examined preferences for receiving WGS results. METHODS: A US nationally representative survey (n = 410 adults) was used to rank preferences for who decides (an expert panel, your doctor, you) which WGS results are reported. We estimated the value of information about variants with varying levels of clinical usefulness by using willingness to pay contingent valuation questions. RESULTS: The results were as follows: 43% preferred to decide themselves what information is included in the WGS report. 38% (95% confidence interval (CI): 33-43%) would not pay for actionable variants, and 3% (95% CI: 1-5%) would pay more than $1,000. 55% (95% CI: 50-60%) would not pay for variants for which medical treatment is currently unclear, and 7% (95% CI: 5-9%) would pay more than $400. CONCLUSION: Most people prefer to decide what WGS results are reported. Despite valuing actionable information more, some respondents perceive that genetic information could negatively impact them. Preference heterogeneity for WGS information should be considered in the development of policies, particularly to integrate patient preferences with personalized medicine and shared decision making.Genet Med 18 12, 1295-1302.

Symptom onset, diagnosis and management of osteoarthritis.

BACKGROUND: The time between symptom onset and physician diagnosis is a period when people with osteoarthritis can make lifestyle changes to reduce pain, improve function and delay disability. DATA AND METHODS: This study analyses data for a nationally representative sample of 4,565 Canadians aged 20 or older who responded to the Arthritis component of the 2009 Survey on Living with Chronic Diseases in Canada. Descriptive statistics are used to report the prevalence of hip and knee osteoarthritis; the mean age of symptom onset and diagnosis; medication use; and contacts with health professionals during the previous year. RESULTS: Among people with a physician diagnosis of arthritis, 37% reported osteoarthritis. Of these, 70% experienced pain in the hip(s), knee(s), or hip(s) and knee(s). Close to half (48%) of these people experienced symptoms the same year that they were diagnosed; 42% experienced symptoms at least a year before the diagnosis; and 10% experienced symptoms after the diagnosis. Among those who had symptoms before diagnosis, the average time between symptom onset and diagnosis was 7.7 years. INTERPRETATION: Individuals with osteoarthritis may experience symptoms for several years before they obtain a physician diagnosis.

Patient preferences for active ulcerative colitis treatments and fecal microbiota transplantation.

Background: Fecal microbiota transplantation (FMT) is a promising treatment for active ulcerative colitis (UC). Understanding patient preferences can identify treatment features that may impact treatment decisions, improve shared decision-making, and contribute to patient-centered care, which is especially important in the context of novel treatments like FMT. Objectives: We aimed to quantify preferences for active UC treatments, specifically FMT and biologics, and identify patient characteristics associated with different preference patterns. Design: This is a cross-sectional survey study. Methods: We administered a discrete choice experiment (DCE) survey to elicit preferences in a sample of Canadian adults with UC. DCE data were analyzed using a main-effects mixed logit model and used to predict uptake of hypothetical scenarios reflecting alternative combinations of treatment features. Latent class modeling identified heterogeneity in patient preference patterns. Results: Participants' (n = 201) mean age was 47.1 years (SD: 14.5 years), 58% were female, and most (84%) had at least some post-secondary education. Almost half were willing to undergo FMT. When considering treatments for active UC, the most important attributes were chance of remission and severity of rare unknown side effects. All else equal, participants were most likely to uptake treatment that involves oral capsules/pills. Participants in the class with the highest utility for chance of remission were younger, had more severe disease, and 58% indicated that they would be willing to undergo FMT. Conclusion: We identified characteristics of UC patients who are more likely to be interested in FMT using preference elicitation methods. Patient-centered care can be enhanced by knowing which patients are more likely to be interested in FMT, potentially improving satisfaction with and adherence to treatments for active UC to maximize the effectiveness of treatment while considering heterogeneity in patient preferences.

Background and aims: Fecal microbiota transplantation (FMT) is a promising new treatment for active ulcerative colitis. Questions remain around the benefits and risks of FMT treatment for patients with ulcerative colitis. Understanding how patients weigh the treatment features and how treatment features influence their decisions may improve shared decision-making and contribute to patient-centered care, which is especially important for novel treatments like FMT.Using an experimentally designed survey, we aimed to:1. Elicit patient preferences for features of active ulcerative colitis treatments, specifically FMT and biologics; and,2. Identify patient characteristics associated with different preference patterns. Results: We found that younger patients with more severe disease are more likely to try FMT for the treatment of active ulcerative colitis. Oral capsules/pills are the preferred mode of treatment administration. Conclusions: These findings can enhance patient-centered care by characterizing patients who are more likely to be interested in FMT. Aligning treatment with the features that are important to patients can potentially improve satisfaction with and adherence to treatments for active ulcerative colitis to maximize their effectiveness for individual patients.

Patient preferences for active ulcerative colitis treatments and fecal microbiota transplantation.

Are We Capturing the Socioeconomic Burden of Rare Genetic Disease? A Scoping Review of Economic Evaluations and Cost-of-Illness Studies.

BACKGROUND AND OBJECTIVES: Rare diseases have a significant impact on patients, families, the health system, and society. Measuring the socioeconomic burden 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 socioeconomic burden 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 (2011-21). We mapped costs using a previously developed evidence-informed framework of socioeconomic burden 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 diseases, 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.

Impact of an online, individualised, patient reported outcome measures based patient decision aid on patient expectations, decisional regret, satisfaction, and health-related quality-of-life for patients considering total knee arthroplasty: Results from a randomised controlled trial.

RATIONALE: Total knee arthroplasty is a common surgical procedure but not appropriate for all patients with knee osteoarthritis. Patient decision aids (PtDAs) can promote shared decision making and enhance understanding and expectations of procedures among patients, resulting in better discussions between patients and healthcare providers about whether total knee arthroplasty is the most appropriate option. AIMS AND OBJECTIVES: Evaluate impact of an individualised PtDA for osteoarthritis patients considering total knee arthroplasty 1 year after baseline assessment. METHODS: Prospective, randomised controlled trial comparing an intervention arm (IA) and routine care arm (RCA). The IA included an online individualised patient reported outcome measures (PROMs) based PtDA and one-page summary report for the surgeon. We report secondary outcomes from the final assessment: patient expectations, decisional regret, patient satisfaction with outcomes of knee replacement, health-related quality-of-life (HRQOL) and depression. We report changes in HRQOL between baseline and final assessments, study arms, and surgical versus non-surgical patients. Descriptive statistics were used to describe participant characteristics and continuous variables. Dichotomous outcomes (expectations, decisional regret, satisfaction) were analyzed using logistic regression and continuous outcomes (HRQOL, depression) were modelled using linear regression. RESULTS: Overall, 140 participants completed all study assessments (IA: n = 69, RCA: n = 71); n = 108 underwent surgery (IA: n = 49, RCA: n = 59). Regardless of study arm, most participants reported expectations were met, minimal decisional regret, satisfaction with outcomes of knee replacement, and had improvements in HRQOL. While no significant differences in study outcomes were found between study arms, IA results were in the direction hypothesised in favour of the PtDA. CONCLUSIONS: Although we were not able to detect statistically significant benefits associated with implementing this PROMs-based PtDA, there was no apparent negative effect on these outcomes 1 year after baseline. We anticipate there may be benefit to implementing this PtDA earlier in the osteoarthritis care pathway where patients have more opportunities to manage their disease non-surgically.

An online individualised patient decision aid improves the quality of decisions in patients considering total knee arthroplasty in routine care: A randomized controlled trial.

Objective: The objective of this study was to evaluate the effectiveness of an online patient decision aid with individualised potential outcomes of surgery, on the quality of decisions for knee replacement surgery in routine clinical care. Design: A pragmatic Randomized Controlled Trial (RCT) in patients considering total knee replacement at a high-volume orthopedic clinic. Patients were randomized at their routine online pre-surgical assessment to either complete a decision aid or not. At their consultation, those in the intervention arm had a surgeon report summarizing the decision aid results. The primary outcome was decision quality, defined as being knowledgeable and choosing the option that matched informed treatment preferences. Multivariate logistic and linear regression analysis was conducted to consider surgeon level clustering and baseline differences between study arms. Results: Of 163 patients randomized, 155 completed post-surgical surveys and were included in the analysis. The average patient was aged 65 years, obese and had moderate to severe osteoarthritis symptoms at baseline. Patients in the intervention arm had a higher odds of making a quality decision (Odds Ratio â€‹= â€‹2.08, 95% CI: 1.08 to 4.02), predominantly through increased knowledge. Conclusions: This study supports the benefit of a decision aid in combination with a surgeon report to significantly improve decision quality in routine care. While the independent contribution of tailoring the decision aid to patient baseline characteristics and including a surgeon report remains unclear, we demonstrated the feasibility of integrating the decision aid into an online pre-surgical assessment in routine clinical care.

Patient Experiences in the Management of Inflammatory Bowel Disease: A Qualitative Study.

Background: Inflammatory bowel disease (IBD) can lead to substantial impairments of quality-of-life. Clinical guidelines and quality indicators aid physicians in practice but may not reflect the perspectives and experiences of patients with IBD. To address this, the objectives of this study were to understand patient experiences with IBD care and to explore priorities. Methods: Based on a convenience sample of 36 participants, five focus groups were completed at four sites across Canada. Data were analyzed using a deductive thematic analysis approach to assess emergent themes and variability in participants' experiences. Results: Our results are organized by themes of structure, process and outcomes to illustrate common issues with respect to how care is organized in the healthcare system, how patients receive and experience care and how patients perceive the outcomes of their care. Our results frame a health systems quality approach that signal needed improvements in access to care, the need for innovation with respect to virtual medicine, the potential expansion of multidisciplinary team-based care and the importance of addressing the psychosocial dimensions for patients with IBD and their caregivers in order to better deliver patient-centred care. Conclusions: The issues identified have the potential to impact priority areas in the system, IBD care delivery, and how outcomes can be improved by focusing on 'lived experience' and patient-centred care. The differing values and perspectives of all those involved in caring for patients with IBD underscore the importance of good communication with patients, caregivers and family members, as well as staying responsive to evolving needs.

Methods for Conducting Stated Preference Research with Children and Adolescents in Health: A Scoping Review of the Application of Discrete Choice Experiments.

BACKGROUND: Discrete choice experiments (DCEs) are a common method used to describe and quantitatively assess preferences in health applications. Increasingly, DCEs have been used to elicit preferences from children and adolescents and generate evidence to inform policies affecting this population. OBJECTIVES: The aim of this review was to summarize and describe the application of DCEs conducted with children and adolescents and describe author-reported age-specific considerations in design, implementation, and analysis. METHODS: A scoping review was conducted using a 'pearl-growing' technique whereby the reference lists of existing systematic reviews of DCEs were used to identify potential studies conducted with children or adolescents as respondents published between 1990 and 2017. This list was supplemented with an updated electronic search using the same strategy as the initial reviews to identify studies from 2017 to 2020. RESULTS: Of 480 studies identified, 19 were included; topics included vaccines (32%), drugs/medical devices (26%), treatment or health promotion interventions/programs (21%), warning labels on cigarettes/nicotine products (10%), and preferences for physical activity and healthy food choices (10%). The youngest reported age for independent DCE completion was 8 years. Approaches to assessing validity and reliability of choices were consistent with best practices for the conduct of DCEs. Reported age-specific considerations included use of visual aids, age-appropriate language, reducing task complexity and cognitive burden, and exploration of interpretation of willingness-to-pay. CONCLUSION: The number of DCEs conducted with children and adolescents has increased in recent years. Detailed explanation of why reported age-specific considerations were necessary, how they could be used to interpret results, or to understand the appropriateness of this methodology for different age groups was limited. Despite a recognition of the need for special consideration when conducting DCEs in this population, the unique issues in the context of age-specific considerations are largely unexplored, and further research is required. Moving forward, stated preference research conducted with children and adolescents should report in more detail methods of recruitment, results of validity assessments, and provide specific reflection on the extent to which modeled results are consistent with expectations and underlying theory.

The price of whole-genome sequencing may be decreasing, but who will be sequenced?

AIM: Since whole-genome sequencing (WGS) information can have positive and negative personal utility for individuals, we examined predictors of willingness to pay (WTP) for WGS. PATIENTS & METHODS: We surveyed two independent populations: adult patients (n = 203) and college seniors (n = 980). Ordinal logistic regression models were used to characterize the relationship between predictors and WTP. RESULTS: Sex, age, education, income, genomic knowledge and knowing someone who had genetic testing or having had genetic testing done personally were associated with significantly higher WTP for WGS. After controlling for income and education, males were willing to pay more for WGS than females. CONCLUSION: Differences in WTP may impact equity, coverage, affordability and access, and should be anticipated by public dialog about related health policy.

How do women trade-off benefits and risks in chemotherapy treatment decisions based on gene expression profiling for early-stage breast cancer? A discrete choice experiment.

OBJECTIVES: Gene expression profiling (GEP) of tumours informs baseline risk prediction, potentially affecting adjuvant chemotherapy decisions for women with early-stage breast cancer. Since only 15% will experience a recurrence, concerns have been raised about potential harms from overtreatment and high GEP costs in publicly funded healthcare systems. We aimed to estimate preferences and personal utility of GEP testing information and benefit-risk trade-offs in chemotherapy treatment decisions. DESIGN, SETTING AND INTERVENTION: Based on literature review and findings from our qualitative research (focus groups, interviews with patients with breast cancer and medical oncologists), we developed a discrete choice experiment (DCE) survey and administered it via an internet panel. The DCE included 12 choice tasks with 5 attributes and 3 alternatives considering orthogonality, D-efficiency and level balance. PARTICIPANTS: The DCE survey was administered to 1004 Canadian women from the general population. MAIN OUTCOME MEASURES: Preferences were analysed using conditional logit and hierarchical Bayes and evaluated for goodness of fit. We conducted simulation analyses for alternative scenarios. RESULTS: GEP test score indicating likely benefit from chemotherapy was the most important attribute. Doctor's clinical estimate of the risk of cancer returning, trust in your cancer doctor and side effects of chemotherapy (temporary and permanent) were relatively less important but showed significant differences among levels. In the scenario analyses, 78% were likely to choose chemotherapy in a high-risk scenario, 55% in a moderate-risk scenario and 33% in a low-risk scenario, with the other attributes held constant. A high GEP score was more important in influencing the choice of chemotherapy for those at intermediate clinical risk. CONCLUSIONS: GEP testing information influences chemotherapy treatment decisions in early-stage breast cancer and varies depending on clinical risk. Clinicians should be aware of these differences and tailor the use of GEP testing accordingly.

The influence of gene expression profiling on decisional conflict in decision making for early-stage breast cancer chemotherapy.

BACKGROUND: Women with early-stage breast cancer, of whom only 15% will experience a recurrence, are often conflicted or uncertain about taking chemotherapy. Gene expression profiling (GEP) of tumours informs risk prediction, potentially affecting treatment decisions. We examined whether receiving a GEP test score reduces decisional conflict in chemotherapy treatment decision making. METHODS: A general population sample of 200 women completed the decisional conflict scale (DCS) at baseline (no GEP test score scenario) and after (scenario with GEP test score added) completing a discrete choice experiment survey for early-stage breast cancer chemotherapy. We scaled the 16-item DCS total scores and subscores from 0 to 100 and calculated means, standard deviations and change in scores, with significance (p < 0.05) based on matched pairs t-tests. RESULTS: We identified five respondent subgroups based on preferred treatment option; almost 40% did not change their chemotherapy decision after receiving GEP testing information. Total score and all subscores (uncertainty, informed, values clarity, support, and effective decision) decreased significantly in the respondent subgroup who were unsure about taking chemotherapy initially but changed to no chemotherapy (n =33). In the subgroup of respondents (n = 25) who chose chemotherapy initially but changed to unsure, effective decision subscore increased significantly. In the overall sample, changes in total and all subscores were non-significant. CONCLUSIONS: GEP testing adds value for women initially unsure about chemotherapy treatment with a decrease in decisional conflict. However, for women who are confident about their treatment decisions, GEP testing may not add value. Decisions to request GEP testing should be personalised based on patient preferences.

Consumer familiarity, perspectives and expected value of personalized medicine with a focus on applications in oncology.

AIMS: Knowledge of consumer perspectives of personalized medicine (PM) is limited. Our study assessed consumer perspectives of PM, with a focus on oncology care, to inform industry, clinician and payer stakeholders' programs and policy. MATERIALS & METHODS: A nationally representative survey of 602 US consumers' ≥30 years old explored familiarity, perspectives and expected value of PM. RESULTS: Most (73%) respondents have not heard of 'personalized medicine,' though after understanding the term most (95%) expect PM to have a positive beneft. Consumer's willingness to pay is associated with products' impact on survival, rather than predicting disease risk. If testing indicates consumers are not candidates for oncology therapies, most (84%) would seek a second opinion or want therapy anyway. CONCLUSIONS: Understanding heterogeneity in consumer perspectives of PM can inform program and policy development.

Estimating the Burden of Osteoarthritis to Plan for the Future.

OBJECTIVE: With aging and obesity trends, the incidence and prevalence of osteoarthritis (OA) is expected to rise in Canada, increasing the demand for health resources. Resource planning to meet this increasing need requires estimates of the anticipated number of OA patients. Using administrative data from Alberta, we estimated OA incidence and prevalence rates and examined their sensitivity to alternative case definitions. METHODS: We identified cases in a linked data set spanning 1993 to 2010 (population registry, Discharge Abstract Database, physician claims, Ambulatory Care Classification System, and prescription drug data) using diagnostic codes and drug identification numbers. In the base case, incident cases were captured for patients with an OA diagnostic code for at least 2 physician visits within 2 years or any hospital admission. Seven alternative case definitions were applied and compared. RESULTS: Age- and sex-standardized incidence and prevalence rates were estimated to be 8.6 and 80.3 cases per 1,000 population, respectively, in the base case. Physician claims data alone captured 88% of OA cases. Prevalence rate estimates required 15 years of longitudinal data to plateau. Compared to the base case, estimates are sensitive to alternative case definitions. CONCLUSION: Administrative databases are a key source for estimating the burden and epidemiologic trends of chronic diseases such as OA in Canada. Despite their limitations, these data provide valuable information for estimating disease burden and planning health services. Estimates of OA are mostly defined through physician claims data and require a long period of longitudinal data.