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Background: Direct estimates of rare disease prevalence from public health surveillance may only be available in a few catchment areas. Understanding variation among observed prevalence can inform estimates of prevalence in other locations. The Muscular Dystrophy Surveillance, Tracking, and Research Network (MD STARnet) conducts population-based surveillance of major muscular dystrophies in selected areas of the United States. We identified sources of variation in prevalence estimates of Duchenne and Becker muscular dystrophy (DBMD) within MD STARnet from published literature and a survey of MD STARnet investigators, then developed a logic model of the relationships between the sources of variation and estimated prevalence. Results: The 17 identified sources of variability fell into four categories: (1) inherent in surveillance systems, (2) particular to rare diseases, (3) particular to medical-records-based surveillance, and (4) resulting from extrapolation. For the sources of uncertainty measured by MD STARnet, we estimated each source's contribution to the total variance in DBMD prevalence. Based on the logic model we fit a multivariable Poisson regression model to 96 age–site–race/ethnicity strata. Age accounted for 74% of the variation between strata, surveillance site for 6%, race/ethnicity for 3%, and 17% remained unexplained. Conclusion: Variation in estimates derived from a non-random sample of states or counties may not be explained by demographic differences alone. Applying these estimates to other populations requires caution. [ABSTRACT FROM AUTHOR]

Background: Although treatment and control of diabetes can prevent complications and reduce morbidity, few data sources exist at the state level for surveillance of diabetes comorbidities and control. Surveys and electronic health records (EHRs) offer different strengths and weaknesses for surveillance of diabetes and major metabolic comorbidities. Data from self-report surveys suffer from cognitive and recall biases, and generally cannot be used for surveillance of undiagnosed cases. EHR data are becoming more readily available, but pose particular challenges for population estimation since patients are not randomly selected, not everyone has the relevant biomarker measurements, and those included tend to cluster geographically. Methods: We analyzed data from the National Health and Nutritional Examination Survey, the Health and Retirement Study, and EHR data from the DARTNet Institute to create state-level adjusted estimates of the prevalence and control of diabetes, and the prevalence and control of hypertension and high cholesterol in the diabetes population, age 50 and over for five states: Alabama, California, Florida, Louisiana, and Massachusetts. Results: The estimates from the two surveys generally aligned well. The EHR data were consistent with the surveys for many measures, but yielded consistently lower estimates of undiagnosed diabetes prevalence, and identified somewhat fewer comorbidities in most states. Conclusions: Despite these limitations, EHRs may be a promising source for diabetes surveillance and assessment of control as the datasets are large and created during the routine delivery of health care. Trial Registration: Not applicable. [ABSTRACT FROM AUTHOR]

Background: Little is known about the effect of drought on all-cause mortality, especially in higher income countries such as the United States. As the frequency and severity of droughts are likely to increase, understanding the connections between drought and mortality becomes increasingly important.Methods: Our exposure variable was an annual cumulative drought severity score based on the 1-month, county-level Standardized Precipitation Evapotranspiration Index. The outcome variables of demographic subgroup-specific all-cause mortality count data per year were obtained from the National Vital Statistics System. Any counts below 10 deaths were censored in that demographic group per county. We modeled county-stratum-year mortality using interval-censored negative binomial regression with county-level random intercepts, for each combined age-race-sex stratum either with or without further stratification by climate regions. Fixed effects meta-regression was used to test the associations between age, race, sex, and region with the drought-mortality regression coefficients. Predictive margins were then calculated from the meta-regression model to estimate larger subgroup (e.g., 'race' or 'sex') associations of drought with mortality.Results: Most of the results were null for associations between drought severity and mortality, across joint strata of race, age, sex and region, but incidence rate ratios (IRRs) for 17 subgroups were significant after accounting for the multiple testing; ten were < 1 indicating a possible protective effect of drought on mortality for that particular subpopulation. The meta-regression indicated heterogeneity in the association of drought with mortality according to race, climate region, and age, but not by sex. Marginal means of the estimated log-incidence rate ratios differed significantly from zero for age groups 25-34, 35-44, 45-54 and 55-64; for the white race group; and for the South, West and Southwest regions, in the analysis that included wet county-years. The margin of the meta-regression model suggested a slightly negative, but not statistically significant, association of drought with same-year mortality in the overall population.Conclusions: There were significant, heterogeneous-direction associations in subpopulation-stratified models, after controlling for multiple comparisons, suggesting that the impacts of drought on mortality may not be monolithic across the United States. Meta-regression identified systematic differences in the associations of drought severity with all-cause mortality according to climate region, race, and age. These findings suggest there may be important contextual differences in the effects of drought severity on mortality, motivating further work focused on local mechanisms. We speculate that some of the estimated negative associations of drought severity with same-year mortality could be consistent with either a protective effect of drought on total mortality in the same year, or with a delayed health effect of drought beyond the same year. Further research is needed to clarify associations of drought with more specific causes of death and with sublethal health outcomes, for specific subpopulations, and considering lagged effects occurring beyond the same year as the drought. [ABSTRACT FROM AUTHOR]