Abstract
Background: For 10 years, the Muscular Dystrophy Surveillance, Tracking, and Research Network (MD STARnet) conducted surveillance for Duchenne and Becker muscular dystrophy (DBMD). We piloted expanding surveillance to other MDs that vary in severity, onset, and sources of care. Methods: Our retrospective surveillance included individuals diagnosed with one of nine eligible MDs before or during the study period (January 2007–December 2011), one or more health encounters, and residence in one of four U.S. sites (Arizona, Colorado, Iowa, or western New York) at any time within the study period. We developed case definitions, surveillance protocols, and software applications for medical record abstraction, clinical review, and data pooling. Potential cases were identified by International Classification of Diseases, Ninth Revision, Clinical Modification (ICD-9-CM) codes 359.0, 359.1, and 359.21 and International Classification of Diseases, Tenth Revision (ICD-10) codes G71.0 and G71.1. Descriptive statistics were compared by MD type. Percentage of MD cases identified by each ICD-9-CM code was calculated. Results: Of 2,862 cases, 32.9% were myotonic, dystrophy 25.8% DBMD, 9.7% facioscapulohumeral MD, and 9.1% limb-girdle MD. Most cases were male (63.6%), non-Hispanic (59.8%), and White (80.2%). About, half of cases were genetically diagnosed in self (39.1%) or family (6.2%). About, half had a family history of MD (48.9%). The hereditary progressive MD code (359.1) was the most common code for identifying eligible cases. The myotonic code (359.21) identified 83.4% of eligible myotonic dystrophy cases (786/943). Conclusions: MD STARnet is the only multisite, population-based active surveillance system available for MD in the United States. Continuing our expanded surveillance will contribute important epidemiologic and health outcome information about several MDs.
| Original language | English (US) |
|---|---|
| Journal | Birth Defects Research |
| DOIs | |
| State | Accepted/In press - Jan 1 2018 |
| Externally published | Yes |
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Keywords
- active surveillance
- Clinical Modification (ICD-9-CM) codes
- International Classification of Diseases
- MD STARnet
- medical record abstraction
- muscular dystrophies
- Ninth Revision
- population-based
ASJC Scopus subject areas
- Pediatrics, Perinatology, and Child Health
- Embryology
- Toxicology
- Developmental Biology
- Health, Toxicology and Mutagenesis
Cite this
Muscular Dystrophy Surveillance, Tracking, and Research Network pilot : Population-based surveillance of major muscular dystrophies at four U.S. sites, 2007–2011. / on behalf of the Muscular Dystrophy Surveillance, Tracking, and Research Network (MD STARnet).
In: Birth Defects Research, 01.01.2018.Research output: Contribution to journal › Article
}
TY - JOUR
T1 - Muscular Dystrophy Surveillance, Tracking, and Research Network pilot
T2 - Population-based surveillance of major muscular dystrophies at four U.S. sites, 2007–2011
AU - on behalf of the Muscular Dystrophy Surveillance, Tracking, and Research Network (MD STARnet)
AU - Do, Thuy Quynh N.
AU - Street, Natalie
AU - Donnelly, Jennifer
AU - Adams, Melissa M.
AU - Cunniff, Christopher M
AU - Fox, Deborah J.
AU - Weinert, Richard O.
AU - Oleszek, Joyce
AU - Romitti, Paul A.
AU - Westfield, Christina P.
AU - Bolen, Julie
PY - 2018/1/1
Y1 - 2018/1/1
N2 - Background: For 10 years, the Muscular Dystrophy Surveillance, Tracking, and Research Network (MD STARnet) conducted surveillance for Duchenne and Becker muscular dystrophy (DBMD). We piloted expanding surveillance to other MDs that vary in severity, onset, and sources of care. Methods: Our retrospective surveillance included individuals diagnosed with one of nine eligible MDs before or during the study period (January 2007–December 2011), one or more health encounters, and residence in one of four U.S. sites (Arizona, Colorado, Iowa, or western New York) at any time within the study period. We developed case definitions, surveillance protocols, and software applications for medical record abstraction, clinical review, and data pooling. Potential cases were identified by International Classification of Diseases, Ninth Revision, Clinical Modification (ICD-9-CM) codes 359.0, 359.1, and 359.21 and International Classification of Diseases, Tenth Revision (ICD-10) codes G71.0 and G71.1. Descriptive statistics were compared by MD type. Percentage of MD cases identified by each ICD-9-CM code was calculated. Results: Of 2,862 cases, 32.9% were myotonic, dystrophy 25.8% DBMD, 9.7% facioscapulohumeral MD, and 9.1% limb-girdle MD. Most cases were male (63.6%), non-Hispanic (59.8%), and White (80.2%). About, half of cases were genetically diagnosed in self (39.1%) or family (6.2%). About, half had a family history of MD (48.9%). The hereditary progressive MD code (359.1) was the most common code for identifying eligible cases. The myotonic code (359.21) identified 83.4% of eligible myotonic dystrophy cases (786/943). Conclusions: MD STARnet is the only multisite, population-based active surveillance system available for MD in the United States. Continuing our expanded surveillance will contribute important epidemiologic and health outcome information about several MDs.
AB - Background: For 10 years, the Muscular Dystrophy Surveillance, Tracking, and Research Network (MD STARnet) conducted surveillance for Duchenne and Becker muscular dystrophy (DBMD). We piloted expanding surveillance to other MDs that vary in severity, onset, and sources of care. Methods: Our retrospective surveillance included individuals diagnosed with one of nine eligible MDs before or during the study period (January 2007–December 2011), one or more health encounters, and residence in one of four U.S. sites (Arizona, Colorado, Iowa, or western New York) at any time within the study period. We developed case definitions, surveillance protocols, and software applications for medical record abstraction, clinical review, and data pooling. Potential cases were identified by International Classification of Diseases, Ninth Revision, Clinical Modification (ICD-9-CM) codes 359.0, 359.1, and 359.21 and International Classification of Diseases, Tenth Revision (ICD-10) codes G71.0 and G71.1. Descriptive statistics were compared by MD type. Percentage of MD cases identified by each ICD-9-CM code was calculated. Results: Of 2,862 cases, 32.9% were myotonic, dystrophy 25.8% DBMD, 9.7% facioscapulohumeral MD, and 9.1% limb-girdle MD. Most cases were male (63.6%), non-Hispanic (59.8%), and White (80.2%). About, half of cases were genetically diagnosed in self (39.1%) or family (6.2%). About, half had a family history of MD (48.9%). The hereditary progressive MD code (359.1) was the most common code for identifying eligible cases. The myotonic code (359.21) identified 83.4% of eligible myotonic dystrophy cases (786/943). Conclusions: MD STARnet is the only multisite, population-based active surveillance system available for MD in the United States. Continuing our expanded surveillance will contribute important epidemiologic and health outcome information about several MDs.
KW - active surveillance
KW - Clinical Modification (ICD-9-CM) codes
KW - International Classification of Diseases
KW - MD STARnet
KW - medical record abstraction
KW - muscular dystrophies
KW - Ninth Revision
KW - population-based
UR - http://www.scopus.com/inward/record.url?scp=85052625958&partnerID=8YFLogxK
UR - http://www.scopus.com/inward/citedby.url?scp=85052625958&partnerID=8YFLogxK
U2 - 10.1002/bdr2.1371
DO - 10.1002/bdr2.1371
M3 - Article
C2 - 30070776
AN - SCOPUS:85052625958
JO - Birth Defects Research
JF - Birth Defects Research
SN - 2472-1727
ER -