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
AU - Fox, Deborah J.
AU - Weinert, Richard O.
AU - Oleszek, Joyce
AU - Romitti, Paul A.
AU - Westfield, Christina P.
AU - Bolen, Julie
N1 - Funding Information:
Work was carried out at each of the MD STARnet sites, with writing completed at the Centers for Disease Control and Prevention (CDC). This publication was supported by the Cooperative Agreement numbers, DD000830, DD000831, DD000832, DD000834, DD000835, DD000836, and DD000837 funded by CDC. The authors appreciate the work of Bailey Hill, for her assistance with data analysis. The authors also acknowledge with appreciation the work of the abstractors, local reviewers, data managers, and other MD STARnet personnel, without whom this article would not be possible.
Funding Information:
Work was carried out at each of the MD STARnet sites, with writing completed at the Centers for Disease Control and Prevention (CDC). This publication was supported by the Cooperative Agreement numbers, DD000830, DD000831, DD000832, DD000834, DD000835, DD000836, and DD000837 funded by CDC. The authors appreciate the work of Bailey Hill, for her assistance with data analysis. The authors also acknowledge with appreciation the work of the abstractors, local reviewers, data managers, and other MD STARnet personnel, without whom this article would not be possible. The findings and conclusions in this report are solely the responsibility of the authors and do not necessarily represent the official views of the Centers for Diseases Control and Prevention, the U.S. Department of Health and Human Services, or the authors' affiliated institutions.
Publisher Copyright:
© 2018 Wiley Periodicals, Inc.
PY - 2018/11/15
Y1 - 2018/11/15
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 - Clinical Modification (ICD-9-CM) codes
KW - International Classification of Diseases
KW - MD STARnet
KW - Ninth Revision
KW - active surveillance
KW - medical record abstraction
KW - muscular dystrophies
KW - population-based
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U2 - 10.1002/bdr2.1371
DO - 10.1002/bdr2.1371
M3 - Article
C2 - 30070776
AN - SCOPUS:85052625958
VL - 110
SP - 1404
EP - 1411
JO - Birth Defects Research
JF - Birth Defects Research
SN - 2472-1727
IS - 19
ER -