People with musculoskeletal pain seek more healthcare than the general population, however little is known about the long-term effect on healthcare use. The aim of this study was to examine the consequences of number of musculoskeletal pain sites on long-term care-seeking and healthcare-related costs and explore how health anxiety influences this relationship.We conducted a Danish population-based longitudinal cohort study of 4883 participants combining self-reported survey data from 2008 with ten-year follow-up data from national health registers. Using a causal inference framework, we examined associations between number of pain sites (range 0–7)/level of health anxiety (high/low level) and face-to-face healthcare contacts/healthcare-related costs. Data were analyzed using negative binomial regression with generalized estimating equations. Regression models were adjusted for sex, age, duration of pain, level of education, comorbidity, personality traits, risk of depression, marital status, physical job exposure, and previous healthcare utilization.For each additional pain site general healthcare contacts (Incidence Rate Ratio (IRR): 1.04 (95% CI: 1.03–1.05)), healthcare-related costs (IRR: 1.06 (95% CI: 1.03–1.08) and musculoskeletal healthcare contacts (IRR: 1.11 (95% CI:1.09–1.14) increased. Those with high levels of health anxiety at baseline had a slightly higher number of general healthcare contacts (IRR 1.06 (1.01–1.11), independent of number of pain sites. However, level of anxiety did not influence the effect of number of pain sites on any healthcare use or cost outcomes.We found evidence for a causal association between increasing number of pain sites and greater healthcare use and cost, and high levels of health anxiety did not increase the strength of this association. This suggests that number of pain sites could be a potential target for biopsychosocial interventions in order to reduce the need for future care-seeking.The online version contains supplementary material available at 10.1186/s12913-021-06994-0. Musculoskeletal pain is common among adults and one of the most common reasons for care-seeking , 2. PeopThe choice to seek care due to pain has been conceptualized by different theories, with the dominant conceptual framework of health services use being the ‘health behavioral model’ . This moHealthcare utilization due to pain and psychological factors, such as health anxiety, has been studied for some years but most research has been undertaken using retrospective or cross-sectional designs of short-term care-seeking from specific healthcare professions or settings  based on self-reported data, with inherent risks of lag time and recall bias , 21. TheThe aim of this study was to examine the consequences of number of musculoskeletal pain sites on long-term care-seeking and healthcare-related costs and explore whether health anxiety influences this relationship. Our hypotheses were that (i) increasing number of pain sites would result in higher total healthcare utilization and total healthcare-related costs over a subsequent 10-year period, (ii) this relationship would be stronger for people with high levels of health anxiety.n = 29), died during the 10-year follow-up (n = 153) or were living abroad for more than 2 years of the follow-up period (n = 32). Participants were between 17 and 65 years of age at baseline. Using validated scales, the questionnaire covered a wide range of information on individual, psychosocial and work-related factors. The following variables were extracted from those baseline questionnaires.This is a population-based, longitudinal cohort study. In February 2008, web-based and postal questionnaires were sent out to 8517 working age people, registered at the same medical center in the Danish city of Odder staffed by eight General Practitioners (GPs). Overall, 5097 people returned the baseline questionnaire, of whom 4883 were eligible for analysis in the current study. Detailed descriptions of the responders and non-responders have been published elsewhere . In the The number of pain sites at baseline was measured using the first part of the pain module of the Standard Evaluation Questionnaire (SEQ) . ParticiHealth anxiety/Illness worry at baseline was measured using the Whiteley-7 Index . Those sTo capture the five basic dimensions of personality  we used the five-factor model of personality traits, derived from 20 items in the Mini International Personality Item Pool (IPIP) which is a validated tool for measuring personality traits , 29. In Risk of depression at baseline was measured using six items (SCL_DEP6) from the Symptom Checklist 90-items. These items are included in the Common Mental Disorder Questionnaire (CMDQ). The CMDQ was developed and validated as a case-finding instrument in primary care . This quDuration of pain at baseline was measured with a single question item from part 4D of SEQ . ParticiUsing the civil registration number uniquely assigned to each resident of Denmark, baseline data was linked to Danish health and social registers to extract data for the subsequent 10-year period (2008–2017) , 31. NumThe National Health Insurance Service Register (NHSR) was established in 1990 and contains information about all fully or partially public funded primary healthcare services. The NHSR contains information about the healthcare provider, public expenditure for each contact and the type of service provided based on the week reimbursement was claimed for , 36. TheThe National Patient Register (NPR) is the central register for recording activity in the Danish secondary healthcare system. The NPR contains information on hospital admissions since 1978 and all outpatient hospital contacts since 1994. Information in the NPS includes the date and time for hospital arrival and departure, type of contact, diagnosis, treatment and tests. Registration in the NPS is based on the Healthcare Classification System  and diagThe Diagnoses Related Group (DRG) Grouped National Patient Register covers data about costs related to in- and out-patient contacts in the NPR. DRG-grouped NPR data itemize payment instances and rates in the Danish healthcare system. Estimated grouped rates for each hospital service are based on the average costs for all hospitals in Denmark and are used for government payments to Danish hospitals. The payment rates for DRG-grouped NPR items are re-evaluated each year.The Rehabilitation According to “The Danish Act of Health §140” register (Rehab Register) was established in 2007. According to Danish law, after hospital admission or outpatient encounters, patients can be referred to publicly funded physiotherapy and occupational therapy rehabilitation if medically indicated. The Rehab Register contains information about contact dates and number of contacts during this type of rehabilitation.Data on sex and age was obtained from the Danish Civil Registration System , 31, 38.Highest achieved level of education at each follow-up year was obtained from The Danish Education Register . ClassifComorbidity was obtained by applying an updated version of Charlson comorbidity index to ICD10 diagnostic codes in NPR . The ChaPhysical lower body occupational job exposure information was obtained by linking the Danish version of the International Standard Classification of Occupations (D-ISCO 88) job titles from the Register-Based Labour Force Statistics , 45 to tMarital status and number of resident children under the age of eighteen was obtained from the Danish Civil Registration System . We combUse of healthcare the last 2 years before baseline was derived by applying the procedure and algorithm for the dependent variable Table ) on 2006For the purpose of this study, two categories of outcomes were derived from these healthcare registers: the number of face-to-face contacts, and healthcare costs. Number of face-to-face contacts were derived separately for musculoskeletal healthcare (MSK-contacts) and for healthcare contacts for any reason (All-contacts). These two outcomes were derived by counting healthcare contacts per participant for each follow-up year (2008 to 2017) in the HSR, NPR and Rehab registers. Details for this procedure are displayed in Table www.dagitty.net) [This study is based on a causal inference framework –53. Eachtty.net)  (Appenditty.net) , 55.To describe the sample, we used frequencies and percentages for categorical and dichotomous variables and means/medians with standard deviations or 25th /75th percentiles for continuous variables. To understand the relationship between key variables, the correlation between number of pain sites and health anxiety, as well as between the outcomes of costs and counts , were tested with Spearman’s correlation coefficients with 95% CI estimated using bootstrapping methods with 100 repetitions. Each hypothesis was tested via a negative binomial distribution regression model using Generalized Estimating Equations (GEE) to account for multiple observations on the same person over the study time periods and right skewed zero inflated count data . We calculated adjusted incidence rate ratios (IRR) for the total effect between the independent variable  and healthcare visits or related costs with 95% CI. We tested for an interaction between number of pain sites and health anxiety in each outcome model. The choice of variable, and hence adjustment for each hypothesis test, was informed by theoretical considerations and DAGs  and some D-ISCO 88 codes were missing for one or more follow-up years. This was managed with chained multiple imputation techniques imputing ten datasets to account for the uncertainty in the imputation. As a sensitivity analysis, estimates from the primary regression analysis on multiple imputation data were compared with estimates from a full case analysis on non-imputed data. Only results based on multiple imputation data will be presented. Chained multiple imputation was applied on 1187 cases missing baseline variables or physical job exposure. Twenty-seven percent of these cases had missing values on one variable, 49% on two variables and 24% on three or more variables. Most missing values were on physical job exposure (54%).All statistics were performed using STATA . As advised by Bland and Altman , two-sidn = 1669). The vast majority of participants (95%) had no comorbidity at baseline. At baseline, 46% of the participants were living with a partner and had children residing with them. Most of the participants had low level of physical job exposure at baseline (Lower body: 87%. Upper body: 72%).The characteristics of included participants are presented in Table Women and participants above 50 years of age at baseline had higher median number, total All-contacts, total ALL-costs and total MSK-contacts compared to men and participants < 50 years of age at baseline. The median number of total ALL-contacts, total ALL-costs and total MSK-contacts increased with increasing number pain sites, high level of health anxiety, duration of pain and high risk of depression. For level of co-morbidity, educational level, marital status and physical job exposure this pattern was inconsistent , between MSK-contacts and All-costs was 0.44 (95% CI 0.43–0.45), and between All-contacts and All-costs was 0.85 (95% CI 0.84–0.85). Correlation between the number of pain sites and health anxiety was 0.39 (95% CI 0.38–0.40).Based on the theoretical model displayed in Appendix C ) and All-costs (IRR: 1.09 (95% CI: 0.99–1.20)) Table . This inAllowing for different effect of sex and age in the adjustment model revealed that females below the age of 50 had more All-contacts and higher All-costs than age-matched males. This difference was not evident for MSK-contacts Fig. . EstimatBased on a causal inference framework, this population-based cohort study found that for every additional pain site a participant reported at baseline, over the subsequent 10 years their healthcare-contacts for any reason, healthcare-related costs and musculoskeletal-related healthcare contacts increased. Non-overlapping confidence intervals for these estimates show that this increase was highest for musculoskeletal-related contacts (11%). Independently of number of pain sites, adults with high level of health anxiety at baseline had more healthcare-contact and higher healthcare-related costs compared to people with low level health anxiety, but this association was weak. Additionally, we found no evidence that level of health anxiety (high vs low) influenced the effect of number of pain sites on healthcare utilization. However, we did find that females below the age of 50 had more healthcare-contacts for any reason and higher healthcare-related costs than age-matched males.This study has several strengths. The prospective, population-based cohort design ensured that information about exposures and co-variables were collected prior to, and independently, of the outcome, which prevents differential misclassification. By linking national health registers and administrative outcomes data to self-reported baseline survey data via personal identification numbers, we insured person-level complete data linkage of healthcare contacts and costs in both the primary and the secondary healthcare sector over a ten-year follow-up period. The validity and completeness of Danish national healthcare register data for number of contacts is considered high , 34, 57.However, some limitations should be noted. As the NHSR database does not contain information on diagnostic coding, MSK-contacts in primary health were based on information about professional groups e.g. physiotherapists, chiropractors and orthopedic surgeons, where their scope of practice indicates that any consultations likely relate to musculoskeletal complaints. For GP contacts, we developed an algorithm to identify MSK-specific contacts , but number of pain sites show quite similar associations between All-contacts and All-costs. This is understandable given the high correlation (0.85) between these outcomes and indicate that general healthcare utilization is similarly estimated by either method.Previous studies have found that health reassurance-seeking is prevalent among individuals with high levels of health anxiety and they tend to make stronger requests to healthcare professionals for expensive diagnostic tests and unnecessary treatments . We founIn designing this study, we took the position that increasing number of pain sites leads to more healthcare-seeking and costs and that this mechanism works through pathways of factors, such as catastrophizing, fear avoidance beliefs and health-related quality of life. This position aligns with behavioral models, e.g. the ‘fear avoidance model’  or ‘the In most Western countries, care-seeking has gained increasing attention as the prevalence of pain and healthcare-related costs has increased during the past two decades , 75. OneThe purpose of this study was not to analyse if increased healthcare use with increased pain sites is helpful, or a result of healthcare overuse or if it is guideline-adherent and evidence based. However, such topics are important for future projects. Instead these results provide insight into the relationship between number of musculoskeletal pain sites, health anxiety and healthcare utilization and highlights factors that may contribute to non-guideline adherent clinical pathways.Our findings show that increasing number of pain sites is associated with higher number of general healthcare contacts, higher healthcare-related costs and higher number of musculoskeletal healthcare contacts over a subsequent ten-year period. We found a weak association between health anxiety and higher number of general healthcare contacts and no evidence that level of health anxiety influences the effect of number of pain sites on healthcare utilization outcomes. In this context, our results add knowledge about drivers of care-seeking and may assist healthcare professionals in formulating patient communication and clinical decision-making in order to optimize healthcare utilization. This study is also a step towards better understanding of a population of patients that might not benefit from current clinical pathways and the organization of healthcare systems in most Western countries. The comorbid nature of pain characterized by multiple pain sites calls for comprehensive collaboration across disciplines which can be a challenge within the silo-organization of most healthcare systems and healthcare sectors. In order better embrace this population of patients in the healthcare system and avoid healthcare overuse, we need more knowledge about the healthcare pain management trajectories across sectors and disciplines. Such knowledge could potentially highlight management gaps or specific patient groups in high risk of non-guideline-adherent clinical pathways. Another important knowledge gap for future research projects of health care service use is the perspective of the health care user. Such knowledge could also guide healthcare providers in their communication with people with multi-site pain in order optimize patient-centered healthcare pain management.Additional file 1.Additional file 2.Additional file 3.


Can physicians accurately estimate a patient’s out-of-pocket expenses if they are given all the necessary information about a drug’s price and the patient’s insurance plan?In this survey study of 371 primary care physicians, gastroenterologists, and rheumatologists, only 21% could accurately estimate out-of-pocket drug costs using information about the drug’s price and an insurance plan’s cost-sharing mechanisms, including deductibles, copays, coinsurance, and out-of-pocket maximums.These findings suggest that few physicians are able to estimate out-of-pocket costs accurately enough to have informed conversations about financial trade-offs with their patients. This survey study examines whether physicians can accurately estimate out-of-pocket expenses when they are given all of the necessary information about a drug’s price and a patient’s insurance plan. One-third of US residents have trouble paying their medical bills. They often turn to their physicians for help navigating health costs and insurance coverage.To determine whether physicians can accurately estimate out-of-pocket expenses when they are given all of the necessary information about a drug’s price and a patient’s insurance plan.This national mail-in survey used a random sample of US physicians. The survey was sent to 900 outpatient physicians . Physicians were excluded if they were in training, worked primarily for the Veterans Administration or Indian Health Service, were retired, or reported 0% outpatient clinical effort. Analyses were performed from July to December 2020.In a hypothetical vignette, a patient was prescribed a new drug costing $1000/month without insurance. A summary of her private insurance information was provided, including the plan’s deductible, coinsurance rates, copays, and out-of-pocket maximum. Physicians were asked to estimate the drug’s out-of-pocket cost at 4 time points between January and December, using the plan’s 4 types of cost-sharing: (1) deductibles, (2) coinsurance, (3) copays, and (4) out-of-pocket maximums. Multivariate linear regression was used to assess differences in performance by specialty, adjusting for attitudes toward cost conversations, demographics, and clinical characteristics.The response rate was 45% (405 of 900) and 371 respondents met inclusion criteria. Among the respondents included in this study, 59% (n = 220) identified as male, 23% (n = 84) as Asian, 3% (n = 12) as Black, 6% (n = 24) as Hispanic, and 58% (n = 216) as White; 30% (n = 112) were primary care physicians, 35% (n = 128) were gastroenterologists, and 35% (n = 131) were rheumatologists; and the mean (SD) age was 49 (10) years. Overall, 52% of physicians (n = 192) accurately estimated costs before the deductible was met, 62% (n = 228) accurately used coinsurance information, 61% (n = 224) accurately used copay information, and 57% (n = 210) accurately estimated costs once the out-of-pocket maximum was met. Only 21% (n = 78) of physicians answered all 4 questions correctly. Ability to estimate out-of-pocket costs was not associated with specialty, attitudes toward cost conversations, or clinic characteristics.This survey study found that many US physicians have difficulty estimating out-of-pocket costs, even when they have access to their patients' insurance plans. The mechanics involved in calculating real-time out-of-pocket costs are complex. These findings suggest that increased price transparency and simpler insurance cost-sharing mechanisms are needed to enable informed cost conversations at the point of prescribing. Later in the year, once she has met her deductible, she likely pays either a flat fee (copay) or a percentage of each drug’s cost (coinsurance). If she incurs enough medical expenses, she may reach her out-of-pocket maximum before the end of the year. Once she meets this limit, she pays nothing at all for prescription drugs for the rest of the year. Such cost-sharing mechanisms exist to address moral hazard and prevent overconsumption of medical resources, but their complexity often confuses patients,13 who turn to their clinicians for assistance.20 Although patients may not expect clinicians to have all the answers, they do report hoping for changes in prescription patterns or referrals to pharmaceutical assistance programs when out-of-pocket costs are projected to be high.22To make matters worse, out-of-pocket costs are often difficult to predict, leaving patients and clinicians unable to account for costs in their medical decisions.27 Cost conversations have the potential to improve relationships between patients and physicians, decrease out-of-pocket costs, and improve outcomes, especially when those costs are known ahead of time.29 Although prior work has shown that physicians agree that discussing costs is important,22 it is still unclear whether physicians understand insurance cost-sharing mechanisms well enough to have informed cost conversations with their patients.30 In this study, we report the results of a survey aimed at determining whether physicians can accurately estimate a patient’s out-of-pocket expenses when they are given all the necessary information about a medication’s price and a patient’s insurance plan.Physician experts and patient advocacy groups have recommended that communication about the financial trade-offs of potential treatment options become part of routine clinical practice.We surveyed 300 primary care physicians , 300 gastroenterologists, and 300 rheumatologists who were randomly selected from the American Medical Association (AMA) Masterfile of all licensed physicians in the US. Data were obtained from Redi-Data, an AMA authorized database licensee. The confidential survey was sent by mail to each physician’s home address in December 2019 with a $5 cash incentive. Survey recipients were told that the goal of the survey was “to understand how physicians think about discussing out-of-pocket costs with their patients” and that their answers would be anonymous. A second wave of surveys was sent to nonrespondents in March 2020. We excluded physicians who were in training, worked primarily for the Veterans Administration or Indian Health Service, were retired, or reported 0% outpatient clinical effort.We entered survey responses into an electronic database, using Qualtrics software. Each survey was entered twice, by 2 different people. Any discrepancies between duplicate entries were reconciled by referencing the original survey.AAPOR) reporting guideline for calculating response rate.The Duke University institutional review board approved the study protocol and deemed that voluntary completion of the questionnaire reflected consent to participate in the study. This study followed the American Association for Public Opinion Research . The full version of the survey that was sent to primary care physicians is shown in the eFigure in the We collected information on demographics  and practice characteristics . Respondents could write in other gender and race categories if those included in the survey were not applicable to them.31Physicians were given 9 statements assessing their attitudes toward having cost conversations with patients. Response options used a 6-point Likert scale ranging from strongly disagree to strongly agree. These statements addressed their thoughts on the cost conversations themselves , barriers to effective cost conversations , and perception of patients’ expectations regarding cost conversations. Statements were adapted from a 2003 study conducted by Alexander and colleagues.Physicians’ ability to estimate out-of-pocket costs was assessed using a short patient vignette . In the Four questions asked physicians to estimate how much the patient would have to pay out of pocket at 4 time points between January and December , years of experience, composite categories of attitudes toward cost conversations, percentage outpatient time, academic affiliation, and access to cost information via the EHR. We did not include information on patient payer mix because of a large number of missing data.P < .05 as a measure of statistical significance. All statistical analyses were performed with Stata software version 16.0 (StataCorp) from July to December 2020. A prespecified statistical analysis plan was preregistered with Open Science Framework.32All tests were 2-sided and used 33 After excluding 34 physicians who reported 0% outpatient clinical time, being retired, or working for the Veterans Administration, we included 371 physicians in our analysis. Of these, 220 physicians (59%) identified as male, 84 (23%) as Asian, 12 (3%) as Black, 24 (6%) as Hispanic, 216 (58%) as White; and the mean (SD) age was 49 (10) years. The mean (SD) years of experience was 22 (10). Specialties were fairly equally represented, with 112 primary care physicians (30%), 128 gastroenterologists (35%), and 131 rheumatologists (35%). Physicians spent a mean (SD) of 83% (22%) of their time in the outpatient setting. They estimated that a mean (SD) of 72% (24%) of their patients were enrolled in Medicare or private insurance. 352 physicians used an EHR (95%), but only 92 (25%) had access to any out-of-pocket cost or coverage information through an EHR.Of the 900 physicians surveyed, 22 were unreachable by mail. Of the remaining 878 physicians, 405 returned completed questionnaires, for an overall response rate of 45% (405 of 900).Among all survey respondents, 354 (87%) responded to the first wave of the study and 51 (13%) responded to the second wave, which was sent at the beginning of the COVID pandemic in the US. Surveys were anonymous and deidentified, so we were unable to compare the characteristics of physicians who responded to the survey with those of physicians who did not respond.In exploratory factor analysis, we reduced questions on attitudes toward cost conversations to 3 composite categories, labeled as (1) duty, (2) perceived barriers, and (3) perceived expectations (see eTable 2 in the The mean (SD) percentage of cost-sharing questions answered correctly was 58% (33%), indicating that, on average, physicians answered approximately 2 out of the 4 questions correctly. Overall, 192 physicians (52%) accurately estimated the tier 4 drug’s cost before the deductible was met; 228 (62%) accurately used coinsurance information to estimate the drug’s cost once the deductible was met; 224 (61%) accurately used drug copay information to estimate the cost of 3 other tier 1 drugs; and 210 (57%) accurately estimated the tier 4 drug’s cost once the out-of-pocket maximum was met . Only 78P = .003). Access to cost or coverage information in the EHR was associated with improved ability to estimate out-of-pocket costs . After adjustment in multivariate analysis, accurate out-of-pocket cost estimations remained associated with access to cost or coverage information in the EHR   .In the sensitivity analysis that defined responses as correct if they fell within a prespecified range  provided inaccurate estimates for at least one aspect of drug-related cost-sharing obligations. Even though a substantial proportion of physicians responding to our survey agreed that they have a responsibility to engage in cost conversations, this view was not associated with improved ability to estimate drug-related out-of-pocket costs.As drug costs rise and insurance plans increasingly rely on cost-sharing strategies such as deductibles and coinsurance,38 or may even be counterproductive if, for example, a physician provides inaccurate information regarding the price of a drug. Future studies should evaluate whether physicians have similar difficulties estimating the costs of other common medical services, including procedures and specialty visits.Physicians’ difficulties estimating drug-related out-of-pocket costs should not be surprising. After all, health care accounting is not part of the medical school curriculum. Moreover, it can be difficult for physicians to keep up with the ever-changing cost-sharing strategies used by insurance plans. As a result, when cost conversations do occur, they are often unproductive40 Our findings suggest that health systems should not rely solely on physicians to calculate and discuss costs with patients. Even if physicians did have a better understanding of insurance coverage, they are unlikely to have the spare time to calculate their patients’ expected costs. Outpatient clinicians already spend a considerable amount of time engaging in shared decision-making about a variety of prevention- and treatment-related issues, especially in the primary care setting.41 Instead, other solutions are needed to help patients anticipate the financial burden of their medical care. In the clinic, pharmacists, social workers, or financial advisors could answer insurance coverage questions before or after visits and help patients apply for copay assistance. Insurance carriers could also make cost estimator tools available to patients online. These tools already exist for some insurance plans, but they have had very poor uptake,44 perhaps in part because patients who use them still have to contact their clinicians to request alternative medications or procedures when costs are too high. Better yet, insurance companies could simplify their cost-sharing mechanisms, especially as evidence grows that patients are bad at choosing insurance plans that provide the most coverage for the least amount of money.45 Finally, insurance companies and health systems could work together to make adjudicated, out-of-pocket costs available in the EHR at the point of prescription. In fact, the Centers for Medicare and Medicaid Services recently began requiring that all Medicare Part D plans adopt out-of-pocket cost estimators and make them compatible with at least one EHR.46 These tools have never been evaluated, however, so it is unknown whether they are accurate, what the optimal EHR design is, and how they affect prescribing patterns and patient outcomes.Prior studies have shown that accurate out-of-pocket cost information can improve patient decision-making.Our study provides some hope that an EHR-based cost estimator could improve physician awareness of drug costs. Although we did not ask physicians to specify the details of their EHRs, those who reported having access to some information about out-of-pocket costs performed better than those who had no information. One possible explanation for this finding is that price transparency, however basic it may be, increases physicians’ awareness of costs and cost-sharing obligations. If that is the case, then point-of-care drug-related out-of-pocket cost information embedded in the EHR could increase the frequency and effectiveness of cost conversations, thereby improving patients’ decision-making capabilities.This study had some limitations. It was limited by a response rate that was likely reduced because of COVID-19–related shutdowns in March 2020. Our results may therefore be affected by nonresponse bias. As the survey was anonymous, we could not evaluate for differences in characteristics between physicians who responded to the survey and physicians who did not respond. It is plausible that physicians who had difficulty answering our questions were less likely to respond to the survey, thus making our results an overestimate of physicians’ ability to accurately estimate out-of-pocket costs. Second, some physicians may have gotten the cost-sharing questions wrong because they did not take the time to do the math needed to answer the questions. If this did happen, it is likely a realistic illustration of the way these physicians approach cost conversations in the clinic. Third, we did not assess physician knowledge of available financial resources, such as pharmaceutical assistance programs and drug rebates. Future studies should evaluate the strategies that patients and clinicians use to alleviate out-of-pocket cost burdens, and how those strategies change depending on patient and clinician understanding of insurance cost-sharing mechanisms. Finally, this survey was targeted at internists and family medicine physicians, so our results are not generalizable to other specialties. Future research should explore understanding of insurance coverage and cost-sharing mechanisms among physicians in other specialties.This survey study found that most physicians agree with the importance of discussing costs with their patients but that few are able to accurately estimate out-of-pocket costs well enough to lead informed conversations about financial trade-offs with their patients. These findings suggest that increased price transparency and simpler insurance cost-sharing mechanisms are needed to enable informed cost conversations at the point of prescribing.


Do clinician practices game pay-for-performance programs by selectively reporting measures on which they already perform well, and does mandating public reporting on patient experience measures improve care?In this cross-sectional analysis of patient experience data from Consumer Assessment of Healthcare Providers and Systems (CAHPS) surveys, practices were more likely to voluntarily include CAHPS measures in a Medicare pay-for-performance program when they previously scored higher on these measures. However, mandatory public reporting of CAHPS measures was not associated with improved patient experiences with care.These findings support calls to end voluntary measure selection in public reporting and pay-for-performance programs, including Medicare’s Merit-Based Incentive Payment System, but also suggest that requiring practices to report on patient experiences may not produce gains. This cross-sectional study examines health care practices’ selection of patient experience measures for quality scoring under the pay-for-performance program and the association between mandated public reporting and performance on those measures within precursor programs of the Medicare Merit-Based Incentive Payment System. Medicare’s Merit-Based Incentive Payment System (MIPS), a public reporting and pay-for-performance program, adjusts clinician payments based on publicly reported measures that are chosen primarily by clinicians or their practices. However, measure selection raises concerns that practices could earn bonuses or avoid penalties by selecting measures on which they already perform well, rather than by improving care—a form of gaming. This has prompted calls for mandatory reporting on a smaller set of measures including patient experiences.To examine (1) practices’ selection of Consumer Assessment of Healthcare Providers and Systems (CAHPS) patient experience measures for quality scoring under the pay-for-performance program and (2) the association between mandated public reporting on CAHPS measures and performance on those measures within precursor programs of the MIPS.This cross-sectional study included 2 analyses. The first analysis examined the association between the baseline CAHPS scores of large practices (≥100 clinicians) and practices’ selection of these measures for quality scoring under a pay-for-performance program up to 2 years later. The second analysis examined changes in patient experiences associated with a requirement that large practices publicly report CAHPS measures starting in 2014. A difference-in-differences analysis of 2012 to 2017 fee-for-service Medicare CAHPS data was conducted to compare changes in patient experiences between large practices (111-150 clinicians) that became subject to this reporting mandate and smaller unaffected practices (50-89 clinicians). Analyses were conducted between October 1, 2020, and July 30, 2021.The primary outcomes of the 2 analyses were (1) the association of baseline CAHPS scores of large practices with those practices’ selection of those measures for quality scoring under a pay-for-performance program; and (2) changes in patient experiences associated with a requirement that large practices publicly report CAHPS measures starting in 2014.P = .004). After 2 to 3 years of the reporting mandate, patient experiences did not differentially improve in affected vs unaffected practices .Among 301 large practices that publicly reported patient experience measures, the mean (IQR) age of patients at baseline was 71.6 (70.4-73.2 ) years, and 55.8% of patients were women . Large practices in the top vs bottom quintile of patient experience scores at baseline were more likely to voluntarily include these scores in the pay-for-performance program 2 years later (96.3% vs 67.9%), a difference of 28.4 percentage points (95% CI, 9.4-47.5 percentage points; In this cross-sectional study of US physician practices that participated in precursors of the MIPS, large practices were found to select measures on which they were already performing well for a pay-for-performance program, consistent with gaming. However, mandating public reporting was not associated with improved patient experiences. These findings support recommendations to end optional measures in the MIPS but also suggest that public reporting on mandated measures may not improve care. However, allowing practices to select the measures they report raises concerns. First, measure selection undermines a central goal of public reporting: to compare clinicians and practices on a consistent set of measures.9 Second, because scoring in the MIPS is based on relative performance , practices have a strong incentive to select measures on which they expect to perform well relative to other practices.12 Thus, measure selection raises concerns about gaming because practices may be able to earn bonuses or avoid penalties by choosing measures on which they already score well, rather than by investing time and resources to provide better care.13Performance in the MIPS is assessed primarily from measures selected by clinicians or their practices. For example, practices can report any 6 of nearly 300 measures in the quality domain, which accounts for nearly half of the overall performance score.8 Such a change would effectively make public reporting mandatory for a core set of measures and eliminate practices’ ability to select which measures affect payment adjustments. However, it remains unclear whether practices select measures on which they already score well, and if mandating public reporting on patient experience measures may be beneficial.Citing these concerns, the Medicare Payment Advisory Commission recommended replacing the MIPS with a program focused on a smaller set of mandatory performance measures, including patient experiences with care.15 We conducted 2 analyses focused on patient experience measures from the Consumer Assessment of Healthcare Providers and Systems (CAHPS) survey. First, we studied measure selection among large practices (≥100 clinicians), which were required to publicly report CAHPS measures starting in 2014 but could voluntarily include these measures in a separate pay-for-performance program.19 We examined the association between practices’ baseline CAHPS scores and subsequent selection of these measures for quality scoring in the pay-for-performance program. Second, we used the introduction of the CAHPS reporting mandate for large practices and a difference-in-differences design to examine the relationship between mandatory public reporting and changes in patient experiences with care.20To address these questions, we studied precursors of the MIPS—the Value-Based Payment Modifier (VM) and Physician Quality Reporting System (PQRS)—the configurations of which enabled us to examine practice measure selection and mandatory public reporting.STROBE) reporting guidelines for cohort studies.21 Analyses were conducted between October 1, 2020, and July 30, 2021.This cross-sectional study was approved by the Harvard Medical School Committee on Human Studies, which granted a waiver of informed consent because we analyzed deidentified secondary data. We followed the Strengthening the Reporting of Observational Studies in Epidemiology , a pay-for-performance program, and the Physician Quality Reporting System (PQRS), a public reporting program.20 Under the VM, practices received upward, downward, or neutral payment adjustments based on their performance in 2 areas: quality of care and per-patient spending. Practices were evaluated on a mandatory set of quality and spending measures but could select additional measures, including CAHPS measures, that contributed to an overall quality score.16 A prior study examined the association between pay-for-performance incentives in the VM and performance on the program’s mandatory measures.14 Here, we examined whether practices included CAHPS measures as an optional component of their overall VM quality score.The VM was introduced as a voluntary program in 2013 and was fully phased in as a mandatory program for practices with 10 or more clinicians by 2015.15 Over time, components of the PQRS became mandatory.20 Beginning in 2014, large practices with 100 or more clinicians were required to publicly report CAHPS measures to avoid penalties.19 Because compliance with this reporting mandate grew rapidly after the first year, we omitted 2014 as a transition year and examined changes in patient experiences from 2011 to 2013 to 2015 to 2016. Additional details about the VM and PQRS are in eTable 1 of the The PQRS was introduced in 2007 and initially paid bonuses to practices that voluntarily reported performance measures, a subset of which were displayed online on Physician Compare.16 Therefore, we excluded practices that participated in the MSSP in any year from 2012 (the program’s first year) through 2016. The MIPS replaced the VM and PQRS in 2017 and made public reporting of patient experiences optional for all practices.22Practices that participated as ACOs in the Medicare Shared Savings Program (MSSP) were not initially included in the VM and reported patient experiences through a separate CAHPS for ACOs survey.17 We observed annual practice-level scores for 11 patient experience domains and whether practices elected to include these scores in their overall VM quality score .We analyzed practice-level data using 2014 to 2016 VM Practice Files, which included the CAHPS scores of large practices that reported these measures for the PQRS. These scores were calculated by CMS based on responses to the CAHPS for PQRS survey, which was administered by a third party to individuals with traditional  Medicare. Medicare beneficiaries were sampled from practices where they received most primary care.24 To assess how practice decisions to include CAHPS scores in the VM changed over time as practices learned about their prior performance, we studied a sample of practices that reported CAHPS measures in 2016 (the last year of the VM and PQRS) and started reporting them in 2014 or 2015.Practices were informed of their CAHPS scores after these measures could have contributed to the practice’s annual VM quality score.16 The fee-for-service Medicare CAHPS survey is administered annually to representative samples of community-dwelling Medicare beneficiaries.26 Because the survey is conducted early each year and asks respondents to rate their care over the prior 6 months, we analyzed surveys administered from 2012 to 2014 and 2016 to 2017 to assess patient experiences in 2011 to 2013 and 2015 to 2016, respectively, omitting the 2015 survey pertaining to the 2014 transition year.We analyzed patient-level data from the fee-for-service Medicare CAHPS survey, which is separate from but closely related to the CAHPS for PQRS survey  that accounted for the plurality of the respondent’s office visits with primary care clinicians in that year.In analyses of measure selection, our primary outcome was an indicator that a practice elected to include CAHPS scores in its overall VM quality score. In analyses of mandatory public reporting, we examined patient experience measures from 5 domains of the fee-for-service Medicare CAHPS survey that corresponded to domains from the CAHPS for PQRS survey: rating of primary physician, physician communication, timely access to care, access to specialists, and care coordination  files.30). Race and ethnicity were assessed from the RTI race variable, which classifies Medicare beneficiaries’ race and ethnicity based on Social Security Administration data and an imputation algorithm that identifies additional Hispanic and Asian beneficiaries.31 Variable descriptions are in section 5 in the Analyses of the fee-for-service Medicare CAHPS survey included respondents’ demographic characteristics , health status, and proxies for socioeconomic status . We compared the proportion of practices that included CAHPS scores, measured in the baseline year and up to 2 years later, in their annual VM quality scores, across quintiles of baseline scores. To examine the relationship between baseline scores and future performance, we calculated correlations of practice-level scores across years. Analyses were conducted using SAS statistical software .We conducted a difference-in-differences analysis to assess changes in patient experiences associated with mandatory public reporting. Specifically, we compared changes in patient experiences from a preintervention period (2011-2013) to a postintervention period (2015-2016) between large practices (111-150 clinicians) affected by the reporting mandate and smaller unaffected practices (50-89 clinicians). For each patient experience score, we estimated a linear difference-in-differences model:i,t,k,c,hScore is a score for respondent i in year t who was attributed to practice k and lived in county c and Hospital Referral Region kh; LargePractice indicates that practice k had 111 to 150 clinicians; and (2015 or 2016)t denotes the postintervention period. We adjusted for respondents’ health, demographic, and socioeconomic characteristics (itX), to account for any compositional changes among patients of large vs smaller practices over time. We adjusted for the annual Medicare Advantage rate by county  to control for potential spillovers of the Medicare Advantage program onto fee-for-service Medicare beneficiaries33; year fixed effects (tyear) to control for time trends; and hospital referral region fixed effects (hHRR) to control for time-invariant market factors affecting care for patients across large and smaller practices.where 3 represents the adjusted within-HRR differential change in patient experiences associated with mandatory public reporting for large practices (pooled across HRRs), through 2 to 3 years after the mandate’s introduction. To facilitate interpretation, we reported estimates of β3 scaled by the practice-level standard deviations (SDs) of preintervention period scores (termed effect sizes). We adjusted for survey weights and used robust variance estimation to account for clustering within practices. Section 5 in the P < .05 using 2-sided tests.Thus, β34 First, we estimated differential changes in patient- and practice-level characteristics between large and smaller practices before and after 2014.35 The absence of differential changes supports the assumption that the difference-in-differences model isolates changes in patient experiences associated with public reporting from compositional changes among practices or their patients. Second, we compared preintervention trends in patient experience scores between large and smaller practices. Similar preintervention trends support the assumption that differences in scores between large and smaller practices would have remained constant had the reporting mandate for large practices not been introduced.34To examine practice compliance with the reporting mandate, we assessed rates of public reporting of CAHPS measures among large vs smaller practices during 2015 to 2016, which we compared with rates in 2014. We also conducted 2 tests of assumptions underlying the difference-in-differences design.We conducted 2 sensitivity analyses. First, we examined changes in patient experiences from concurrent CAHPS surveys. Second, we reestimated our difference-in-differences models on a broader sample of survey respondents, whom we attributed to practices based on office visits with primary care clinicians or specialists.Among practices with 100 or more clinicians, 301 publicly reported CAHPS measures in 2016 and started reporting them in 2014 or 2015 (742 practice-years). At baseline (2014 or 2015), these practices had a mean of 431 clinicians and 10 229 patients enrolled in fee-for-service Medicare, among whom the mean age was 71.6 years, 55.8% were women, and 17.8% also received Medicaid . In the subset of practices that first reported CAHPS measures in 2014, the 60 practices in the lowest baseline quintile became less likely to include CAHPS measures by 2016, whereas practices in the highest baseline quintile became more likely to include CAHPS measures by 2016 , underscoring their importance as quality indicators.44However, our results also suggest that mandating public reporting of patient experience measures, as recommended in some MIPS reform proposals,45These findings remain salient amid forthcoming changes to the MIPS. In 2022, CMS will launch the MIPS Value Pathways framework. This framework is intended to reduce reporting burdens by defining core sets of measures, pertaining to specific conditions or specialties, that practices can report. However, MIPS Value Pathways framework may not eliminate gaming, since CMS has indicated that practices will be able to choose which measures (from a given measure set) they report.16 However, practices in the MIPS face similar uncertainty about the set of peers reporting on a given measure, and thus are also likely to rely on assessments of their own past performance relative to other practices when selecting measures.46 Second, findings from our difference-in-differences analyses may not generalize to Medicare beneficiaries living in institutional settings, such as nursing homes, because the fee-for-service Medicare CAHPS survey samples from the community-dwelling Medicare population. Third, our difference-in-differences analyses could have been biased by unmeasured confounders. However, we did not detect meaningful differential changes on observable patient or practice characteristics.Our study had some limitations. First, the extent to which practices strategically selected measures under pay-for-performance was likely diminished by the uncertain payoff  associated with selecting specific measures, since in the VM payment adjustments were based on a composite of performance measures assessed among all practices, and the set of measures and practices varied year-to-year.In precursors of the MIPS, we found that large clinician practices were more likely to voluntarily include patient experience measures in a pay-for-performance program when they previously performed well on these measures. However, mandatory reporting of patient experiences was not associated with improved performance on these measures. These findings underscore concerns about gaming in the MIPS and provide cautionary evidence about proposed reforms to this program.


Aspergillus infection and exclusion of alternative diagnosis, all occurring for more than 3 months. Recently, a rapid, highly sensitive and specific point-of-care lateral flow device (LFD) has been introduced for the detection of Aspergillus-specific immunoglobulin (Ig)G, especially in resource-limited settings where CPA is underdiagnosed and often misdiagnosed as smear-negative pulmonary tuberculosis (PTB). Therefore, in our setting, where tuberculosis (TB) is endemic, exclusion of PTB is an important first step to the diagnosis of CPA. We used the recently published CPA diagnostic criteria for resource-limited settings to identify patients with CPA in our center.Diagnosis of chronic pulmonary aspergillosis (CPA) is based on a combination of clinical symptomatology, compatible chest imaging findings, evidence of Aspergillus-specific IgG/IgM antibody tests. After CPA diagnosis, anti-TB drugs were safely discontinued for all patients, and they were initiated on capsules of itraconazole 200 mg twice daily with good treatment outcomes.Three Ugandan women (45/human immunodeficiency virus (HIV) negative, 53/HIV infected and 18/HIV negative), with a longstanding history of cough, chest pain, weight loss and constitutional symptoms, were clinically and radiologically diagnosed with PTB and empirically treated with an anti-tuberculous regimen despite negative microbiological tests. Repeat sputum Mycobacteria GeneXpert assays were negative for all three patients. On further evaluation, all three patients met the CPA diagnostic criteria with demonstrable thick-walled cavities and fungal balls (aspergilomas) on chest imaging and positive The availability of simple clinical diagnostic criteria for CPA and a LFD have the potential to reduce misdiagnosis of CPA and in turn improve treatment outcomes in resource-limited settings.The online version contains supplementary material available at 10.1186/s13256-021-02721-9. Tuberculosis (TB) is one of the most common causes of death and the leading cause of death from a single infectious agent worldwide. The highest burden is found in Africa and Asia mostly linked to the human immunodeficiency virus (HIV) epidemic in these regions . The 202Aspergillus fumigatus [Chronic pulmonary aspergillosis (CPA) is a slowly progressive and destructive parenchymal lung disease mostly caused by umigatus  and affeumigatus . CPA affumigatus , 7 and 6umigatus . More thumigatus . This grumigatus , 11. Witumigatus , 12, theumigatus . This maumigatus , 15, witAspergillus LFD  [Based on the unified case definition of CPA in resource-limited settings , and the France) . This arA 45-year-old Ugandan woman presented with a 3-month history of coughing up blood-stained sputum associated with left-sided pleuritic chest pain, exacerbated by lying on the same side. This was the first episode of hemoptysis. She reported mild dizziness, but no exertional intolerance, orthopnea or ankle swellings. She had no history of constitutional symptoms. She was a peasant farmer who resided in a rural area and was referred from a peripheral health facility where she had been suspected to have TB and was started on anti-TB therapy for 14 days. She was HIV negative and had no history of previous chronic chest symptoms. She was not a current or ex-smoker of tobacco.2) 98% on ambient air, respiratory rate 24 bpm and temperature 36.8 °C. She had no conjunctivae pallor, jaundice or lymphadenopathy. On chest examination, she was tachypneic with mild respiratory distress. She had reduced chest movements on the left with increased tactile fremitus, dull percussion notes and bronchial breathing in the left infraclavicular region. The trachea was deviated to left with the apex beat displaced to the sixth intercostal space (ICS). There were coarse crepitations on the left axillary region. The remainder of the examinations was unremarkable.On examination, her blood pressure was 120/80 mmHg, pulse rate 80 beats per minute (bpm), peripheral oxygen saturation G/IgM) was positive  for 4 months. Recent CD4 T-counts and viral loads were not known. We did not perform CD4 at that time point. Review of other systems was essentially normal. She was diagnosed and treated four times (6 months each time) for PTB. She had a history of frequent childhood respiratory symptoms of cough and difficulty in breathing, and she was told she suffered from asthma. However, she had no previous documentation and had never used inhaler therapies. She had previously smoke the local tobacco for 2 years and had occasionally used alcohol for 1 year.2 98% on room air. She was wasted with mild pallor of the mucous membrane with digital clubbing. She had no jaundice, edema or lymphadenopathy. On chest examinations, she had mild respiratory distress. She had a flattened right-side chest in the infraclavicular region with reduced chest movements, tracheal deviation to the right and apex beat of the sixth intercostal space (ICS). In the same chest region, there was increased tactile fremitus, a dull percussion note, bronchial breathing and increased vocal resonance. Cardiac and abdominal examinations were normal.On examination, she had a BP of 120/80 mmHg, pulse rate 80 bpm (regular), respiratory rate 22 bpm and SpOAspergillus IgM-IgG LFD .2 94% on room air. She was febrile with a temperature of 38.3 ºC. She was severely pale with no digital clubbing. She had no palpable lymph nodes and no edema. Chest examination revealed a flattened right infraclavicular region with tracheal deviation to the right without displacement of the point of maximum cardiac intensity. There were reduced chest movements on the right hemithorax, with an increased tactile fremitus, dull percussion note and broncho-breathing both posteriorly in the mid-scapular area and in the anterior mammary area. Cardiac examination revealed tachycardia with normal heart sounds.On examination, she had a blood pressure of 110/70 mmHg, respiratory rate 22 breaths per minute, pulse rate 131 beats per minute and SpOChest x-ray showed a homogeneous opacity in the right hemithorax with multiple crowded cysts in the middle and lower lung zones associated with volume loss and ipsilateral mediastinal shift. There was a cavity in the right perihilar region. There were multiple reticulo-nodular opacities scattered throughout the entire right lung field with cystic changes Fig. a. The chMycobacterium tuberculosis. Renal and liver function tests were within normal limits. Aspergillus-specific LFD (IgG-IgM) was positive  after exclusion of an alternative diagnosis. The LDBio Aspergillus LFD has a sensitivity of 92% and a specificity of 98.0% in patients with proven CPA [According to the unified case definition of CPA in resource-limited settings , CPA can setting  showed t setting –21 as thoven CPA . In thisoven CPA . This isoven CPA , 23 and oven CPA .Patients II and III were typical of post-TB lung disease. This was an expected observation in these patients since persistence of pulmonary cavities after successful pulmonary TB treatment is very common  and thesCPA is heavily underdiagnosed and misdiagnosed in resource-limited settings where rapid diagnostics are unavailable . PreviouA well-designed cross-sectional study conducted in a resource-limited setting  with over 70% of the participants being HIV positive reported an overall CPA prevalence of 8.7%, 6.5% among HIV infected and 14.5% among HIV-negative participants . AnotherAs illustrated in the above cases, we have seen patients being subjected to anti-TB medications two or more times even when the sputum test for TB was negative. In addition, obvious cavitary lesions with fungal balls consistent with CPA are treated as active TB lesions because CPA cannot be ruled in. This could be due to the low index of suspicion among clinicians and/or lack of point-of-care fungal diagnostics. We have been able to safely discontinue anti-TBs in patients with negative mycobacterial work-ups who meet the diagnostic criteria for CPA and have commenced them on the appropriate antifungal therapy with an overall good outcome. However, the high cost of itraconazole together with the long duration of treatment is still a major challenge in our country.Aspergillus LFD will change the way CPA is diagnosed in resource-limited settings, reducing misdiagnosis and unnecessary use of anti-TB therapy in this population. There is need to advocate for translational research into developing more fungal diagnostics to address the local disease burden [Based on the experience and the overwhelming success of LFDs in the diagnosis of a sister fungal infection, cryptococcal meningitis, in resource-limited settings , 30, if e burden . In concAdditional file 1: CARE Checklist. File contains a CARE Checklist of information to include when writing a case report.


The COVID-19 pandemic resulted in public health and policy measures to reduce in-person contact and the transmission of the virus. These measures impacted daily life and mental well-being (MWB). The aims of this study were to explore the MWB impacts of COVID-19 on children and assess the associations among perceived changes in physical activity (PA) and sedentary behaviors (SB), with perceived MWB changes, using a mixed-methods approach.A convergent parallel mixed-methods design consisting of an online survey with a convenience sample and interviews was conducted from May through July 2020 with parents/caregivers of kindergarten through 5th graders in the St. Louis region. Survey domains assessed included child MWB, PA, and SB. Interviews were recorded, transcribed, and qualitatively analyzed using a code book developed to elicit themes. Survey data was analyzed with chi-squared tests and logistic regressions. The dependent variable was perceived change in child MWB due to the impact of COVID-19. Independent variables included perceived changes in PA, SB, and child concerns about COVID-19.Sample size consisted of 144 surveys and 16 interviews. Most parents reported a perceived decrease in child MWB (74%), a decrease in child PA (61%), and an increase in child SB (91%). Discontentment with stay-at-home orders and concern about COVID-19 were associated with a perceived decrease in MWB. Children whose PA decreased were 53% less likely to have the same or better MWB (OR 0.47) and children whose outside PA decreased were 72% less likely to have the same or better MWB (OR 0.28). Common qualitative themes included difficulty in adjusting to COVID-19 restrictions due to school closures and lack of socializing, child concerns about family getting sick, and PA benefits for improving MWB.Based on parent perceptions, MWB decreased with COVID-19. Maintained or increased child PA improved the chances MWB would remain the same or improve. Parent interviews provide context to these findings by showing how COVID-19 impacted MWB and the associations between PA and MWB. Understanding protective factors for child MWB during COVID-19 is important to offset negative long-term health outcomes from this ongoing pandemic.The online version contains supplementary material available at 10.1186/s12889-021-11805-6. Beginning in December 2019, the respiratory disease SARS-CoV-2 virus (COVID-19) began spreading around the world. Since then, the World Health Organization has reported over 200 million cases and over 4 million deaths worldwide, with the Centers for Disease Control and Prevention reporting over 36.7 million cases and around 620,000 deaths in the United States (US) as of August 15, 2021 . ChildreThe full impacts of COVID-19 on MWB, PA, and SB are still unknown; however, evidence suggests poor MWB, limited PA, and increased SB in childhood may have potentially long-term and serious health consequences –26. ChilThe COVID-19 pandemic and associated restrictions are ongoing with no clear timeframe for when restrictions will be lifted, making the potential impacts on child MWB, PA, and SB with the subsequent long-term effects on health and well-being particularly concerning. Given this, the aims of this study were 1) to explore how COVID-19 restrictions are impacting child MWB, PA, and SB using qualitative methods, and 2) to assess how perceived changes in PA and SB due to COVID-19 restrictions are associated with perceived changes in MWB using quantitative methods.This was a convergent parallel mixed-methods study to assess the impact of COVID-19 on child MWB and associations with PA and SB. Convergent parallel designs consist of conducting qualitative and quantitative methods independently and concurrently, with results from each of these methods being integrated during the interpretation of findings . The purParticipants for the online survey were recruited using convenience sampling. The survey link was posted on Twitter and Facebook and distributed on Facebook to St. Louis-based neighborhood groups for parents with elementary school children. Sampling methods focused on parents and caregivers in the St. Louis region with children enrolled in kindergarten through 5th grade. The survey was available from late May to early July 2020 for approximately 8 weeks. Participants had the option of providing email addresses. Interviews were conducted independently during this time period within the same population, using a combination of convenience and snowball sampling. Participants were contacted via phone and email. The Institution Review Board of Washington University in St. Louis approved this study with an exempt status.n = 16) were administered via telephone or Zoom and lasted between 20 and 30 min. Parents were provided the consent form and reason for this study prior to the interview. Participants completed the interviews from their homes in privacy. No repeat interviews were conducted. Interviews were conducted until saturation was reached. Interviews were audio recorded for accuracy, de-identified, and then professionally transcribed verbatim. If participants had more than one child, participants were asked to choose one child to answer questions for. Questions asked during the interviews were informed by early COVID-19 research and developed with expertise of the research team. Questions covered similar topics to the online survey, along with more open-ended explorations around how the COVID-19 pandemic impacted their families and children.Participants completed the online survey via Qualtrics. All surveys were anonymous and consisted of a consent page and information regarding the purpose of the study. The online survey was developed for this study .Quantitative data analysis was conducted using SPSS (version 26). The dependent variable was perceived change in MWB. Independent variables included in the analysis were PA behaviors , SB , and COVID-19 concerns . PA and SB were dichotomized into binary variables of ‘same or increased’ and ‘decreased’. COVID-19 concerns were also dichotomized into ‘sometimes true or true’ and ‘not true’. Descriptive statistics, frequency tables, and chi-square analyses were conducted. The McNemar test was run to determine if there were statistically significant differences between each MWB item of the Mood and Feelings questionnaire reported for prior to COVID-19 restrictions compared to during COVID-19 restrictions. For statistically significant correlations, logistic regressions were conducted to determine the association between PA, SB, and COVID-19 concerns with perceived change in MWB. Adjusted models were also run controlling for age and gender.Qualitative analysis of the transcripts began with three rounds of preliminary analysis to develop the qualitative code book. In the first round, we used an open coding method, were a priori codes were developed based on categories of interview questions from the interview guide. In the second round, two research team members reviewed all transcripts and collaborated to add more detailed codes to existing categories. Additional categories for codes were added as needed. In the third round, two research team members used this initial codebook to double-code three transcripts. The two team members then reviewed the coding to identify any gaps in the codebook or differences in coding. Once consensus on coding was established, the codebook was finalized, and a third research team member was trained to use the final codebook. Using the final codebook, each transcript was double-coded by the three members of the research team and reviewed to eliminate discrepancies between reviewers. Coded text was separated out by code to be reviewed for common themes by research team members. Common themes were discussed and agreed upon through consensus. These themes were then compared to the quantitative findings and integrated by identifying convergence and places where qualitative rich text could be used to provide context for quantitative results.The total number of parents surveyed was 245. The final sample consists of the 144 respondents who completed the child Mood and Feelings Questionnaire. The final analytic sample was not significantly different than the total sample of parents surveyed for demographic variables of child age, race, gender, and ethnicity. Table Sixteen qualitative interviews were conducted with parents who had children ranging in age from four to 11 years old, with an average age of 7 (SD = 0.71). Parents reported on a total of 23 elementary school aged children, 13 of which were female. Overall, 87% of households had more than one child. Parents interviewed were mostly female (94%) and married (81%). Parents reported a wide range of jobs from social worker and teacher, to IT consultant, attorney, and business owner. Most reported working from home and a few noted being laid off or furloughed due to the COVID-19 pandemic.Overall, 74% of parents reported their children had worse MWB during COVID-19 restrictions (see Table x2 (2)=8.06, p = 0.005) and concern (x2 (2)=7.20, p = 0.007) about COVID-19 were significantly associated with perceived changes in MWB. Among PA behaviors surveyed, perceived changes in PA (x2 (2)=4.10, p = 0.043), perceived changes in organized sports (x2 (2)=5.61, p = 0.018), and perceived changes in outside play (x2 (2)=6.35, p = 0.012), were significantly associated with perceived changes in MWB. Among SB, only a perceived change in time spent sitting (x2 (2)=4.65, p = 0.031) was statistically significant.Results from chi-squared and logistic regression analyses are presented in Table Logistic regressions were conducted for all statistically significant correlations. Models were adjusted for age and gender to control for potential confounding of these demographic factors. Overall, outcomes did not change significantly after adjusting for age and gender. However, there was evidence of confounding by age for change in sitting. Therefore, results are presented for both unadjusted and age-adjusted models Table . We founThemes from qualitative interviews provided context to how COVID-19 restrictions impacted children’s MWB and elucidated protective factors which minimized the impact of COVID-19 Table . A promiThe findings of this study suggest COVID-19 restrictions in the spring and early summer of 2020 may have impacted child MWB, PA, and SB. Through a mixed-methods approach, we were able to identify potential mechanisms through which these restrictions could impact children and potential ways of mitigating these impacts. While our results are based on parent perceptions around their child’s behavior and well-being during COVID-19 restrictions, this research presents novel findings during an unprecedented time, and provides foundations for further understanding about the role of COVID-19 restrictions on child MWB, PA, and SB.Parents perceived MWB to decrease for all MWB items in the adapted child Mood and Feelings Questionnaire and 74% of parents reported worse child MWB with COVID-19 restrictions. These findings support previous research highlighting the vulnerability of child MWB to the COVID-19 pandemic –35. QualAnother important finding of our study was the association between COVID-19 restrictions and reduced PA. This finding adds to previous research which has found a similar association among adults and children alike –40. ReduWe did not find associations between time spent sitting and changes in MWB after adjusting for age. However, many parents we interviewed, noted that not having the structure of in-person school, along with the added screen time of remote learning, increased SB and in particular time spent sitting, which was often reported to be difficult for children. Parents felt their child’s mood and behavior were negatively impacted, citing how their child often complained of boredom and difficulty concentrating in the remote learning environment. The differences between our quantitative and qualitative findings regarding SB and child MWB, highlight the need for more research into the potential impact of age on child experiences during COVID-19.With ongoing restrictions, efforts should be directed toward increasing socialization remotely, improving communication with children about COVID-19, and finding ways to increase PA and reduce SB during the COVID-19 pandemic. One way many parents identified as an opportunity to increase socialization was through the use of technology. Many parents felt using technology to socialize through multi-player games, video calls, and social media alleviated some of the loneliness their children were feeling due to COVID-19 restrictions. These findings align with research highlighting how technology can be used to maintain social connection during the COVID-19 pandemic , 46. WhiIn discussing child worry and concern about COVID-19, many parents in this sample reported children often worried less after having conversations about restrictions being in place to prevent the transmission of COVID-19 and ways family and friends were protecting themselves from becoming sick. Having these conversations in age-appropriate ways may help alleviate worry and fear some children might experience due to the COVID-19 pandemic and associated restrictions. The Centers for Disease Control and Prevention (CDC) have disseminated guidelines on the importance of communicating with children about COVID-19 and how to speak with children of different ages . Other oOur findings on PA and SB suggest a need for interventions and policies to promote PA and reduce SB during the COVID-19 pandemic and associated restrictions. Outside play was perceived to have the greatest impact on changes in child MWB, which may provide opportunities to promote PA in warmer months but suggests PA may decrease in winter months. While weather is conducive to outdoor activity, family walks or bike rides outside and making time each day for playing in the yard may be options. Additionally, outdoor activity can provide safer conditions for socializing aligning with recommendations for preventing the spread of COVID-19 . If weatThere are a few limitations of this study. First, the convenience sample is small and homogeneous, limiting some opportunities for analysis and generalizability of findings to other populations. While the survey overall had 245 respondents, we were only able to conduct analysis on the 144 respondents who completed the adapted child Mood and Feelings questionnaire. Second, this study was cross-sectional in nature. As such, we can only assess associations between child MWB, PA, and SB during COVID-19 restrictions and cannot make causal inferences. Third, since we only collected data during the months of May through July, our study cannot assess how a longer time spent at home, or how time of year and weather might impact child MWB, PA, and SB. Finally, measures of change in MWB, PA, and SB relied on parent-report and were subject to recall bias. We also did not collect data on weekly minutes of moderate and vigorous PA, limiting our ability to use U.S. physical activity guidelines to assess perceived changes in child PA. Although limited by sample size and parent-report, this study used a mixed methods approach, which provided context and corroboration for quantitative findings from the online survey. Findings of this study also help contribute to new research in the area of COVID-19 and specifically for how COVID-19 restrictions impact children.This study captured perceived changes in MWB, PA and SB at the start of the COVID-19 pandemic through a mixed-methods approach. Based on our findings, COVID-19 had a significant impact on child MWB, with children experiencing worse MWB due to COVID-19 restrictions. According to parents in this study, child PA behaviors decreased, SB increased with COVID-19 restrictions and maintaining or increasing child PA levels and activities acted as a protective factor for child MWB. Our results indicate monitoring child MWB, PA and SB during this ongoing pandemic is critical to identify ways to intervene and prevent potential long-term consequences of poor MWB, decreased PA, and increased SB. Schools and parents should look towards communicating with children about the COVID-19 pandemic to reduce fear and worry, increasing outdoor PA as weather permits, and incorporating short activities to break up SB and maintain PA levels.As the COVID-19 pandemic and associated restrictions continue, ongoing research will be needed to determine effective interventions for child MWB and ways of keeping children active and healthy during these unprecedented times. More information is needed on how COVID-19 restrictions may differentially impact children by race and ethnicity, socioeconomic status, and urbanicity. Additionally, with COVID-19 restrictions ongoing, it will be critical to determine the role of seasonality and duration of COVID-19 restrictions on child MWB, PA, and SB. While this study contributes to an initial understanding of how COVID-19 restrictions may impact child MWB, PA, and SB, more research will be needed to more fully understand both immediate and long-term outcomes for children.Additional file 1: Supplement 1. Online Questionnaire