Neurology
Use of U.S. Administrative Data in Muscular Dystrophy Research: A Scoping Review Anne Lyon Havlik* Anne Havlik Natalie Street Seth Perlman Jamie Zimmerman Catharine Riley
Muscular dystrophies (MD) are a group of hereditary muscle disorders that result in progressive muscle weakness, decreasing mobility, and declining quality of life over time. Administrative healthcare data are a valuable resource that can augment public health surveillance and research, especially when small sample sizes are a challenge. A systematic literature search of Medline, Embase, CINAHL, and Scopus was performed to learn how U.S. administrative data have been used to study MD and identify opportunities and gaps in the research. Key search terms used were related to MD, administrative health data, and healthcare utilization, which retrieved 1,270 bibliographic records; 854 unique abstracts and 53 full-text articles were reviewed. A standardized abstraction form was completed by 2 independent reviewers. Fifty studies met eligibility criteria and were included in the review. Studies used different approaches to MD case ascertainment, resulting in methodological and data quality heterogeneity. Seventeen publications included only claims administrative data, 9 included only inpatient administrative data, no publications included only outpatient administrative data, and 24 publications included at least 2 of these administrative data sources and other sources. Fifteen publications focused on pediatric populations, defined as including individuals less than 21 years of age. Thirty-five publications included populations outside of pediatric ages. Gaps identified include addressing differences in ICD code usage for MD subtypes, characterizing MD in young adult and older populations, and utilizing standardized diagnostic criteria as well as multiple sources of ascertainment. This is the first scoping review of MD research using administrative data. Public health implications may include enhanced evidence-based decision-making for individuals living with MD and their healthcare providers.