Abstract
BackgroundRare conditions can be catastrophic for families and the implications for public health can be substantial. Our study compared basic surveillance through active medical record review with a linked administrative data file to assess the number of cases of two rare conditions, fragile X syndrome (FXS) and muscular dystrophy (MD) in a population.MethodsTwo methods of data collection were used to collect information from five counties comprising two standard metropolitan statistical areas of South Carolina. The passive system relied mostly on health claims data using ICD-9 CM diagnostic codes. The active system relied on a nurse abstracting records from a list of all licensed physicians with specialties in neurology, orthopedics, and genetics.ResultsThere were 141 FXS cases and 348 MD cases that met the case definitions using active surveillance. Additional cases were found for both conditions but they were determined to not be true cases. After linking the actively collected MD and FXS cases to passive datasets, we found that the estimated total numbers of cases were similar to using capture-recapture analysis; the positive predictive values for cases identified in the passive system were 56.6% for MD and 75.7% for FXS.ConclusionsApplying capture-recapture methods to passively collected surveillance data for rare health conditions produced an estimate of the number of true cases that was similar to that obtained through active data collection.
Highlights
Rare conditions can be catastrophic for families and the implications for public health can be substantial
Data collection Two methods of data collection were used for this project: a passive data system whereby data about cases of fragile X syndrome (FXS) and muscular dystrophy (MD) statewide were ascertained through the linkage of multiple administrative data systems and an active data collection system whereby data about cases of FXS and MD were abstracted directly from medical records from medical practices that serve individuals with the two conditions
Both approaches were conducted by state agencies; the passive data system was completed by the Health and Demographics Section of the South Carolina Office of Revenue and Fiscal Affairs (RFA) and the active data collection was completed by Maternal and Child Health Bureau at the South Carolina Department of Health and Environmental Control (DHEC)
Summary
Rare conditions can be catastrophic for families and the implications for public health can be substantial. Our study compared basic surveillance through active medical record review with a linked administrative data file to assess the number of cases of two rare conditions, fragile X syndrome (FXS) and muscular dystrophy (MD) in a population. It is often difficult to effectively and efficiently estimate the number of cases of these rare conditions in a specific area. Some methods, such as reportable condition registries, may require legislation or other state-level policy, while methods such as medical record abstraction are costly and time-intensive. The expectation that linked administrative records, or passive surveillance systems, are reliable sources for estimating the true prevalence for health conditions, has been questioned by many public health professionals and policy makers [4].
Sign-in/Register to view highlights & summary Sign-in/Register to access full text options 
Talk to us
Join us for a 30 min session where you can share your feedback and ask us any queries you have
Schedule a callDisclaimer: All third-party content on this website/platform is and will remain the property of their respective owners and is provided on "as is" basis without any warranties, express or implied. Use of third-party content does not indicate any affiliation, sponsorship with or endorsement by them. Any references to third-party content is to identify the corresponding services and shall be considered fair use under The CopyrightLaw.
