Muscular Dystrophy Surveillance, Tracking, and Research Network pilot: Population-based surveillance of major muscular dystrophies at four US sites, 2007-2011
Muscular Dystrophy Surveillance, Tracking, and Research Network pilot: Population-based surveillance of major muscular dystrophies at four US sites, 2007-2011
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DOI：
10.1002/bdr2.1371
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发表时间：
2018-11-15
影响因子：
2.1
通讯作者：
Bolen, Julie
中科院分区：
文献类型：
--
作者：
Do, ThuyQuynh N.;Street, Natalie;Bolen, Julie
背景方法 十年来，肌营养不良症监测、跟踪和研究网络 (MD STARnet) 对杜氏肌营养不良症和贝克尔肌营养不良症 (DBMD) 进行了监测。我们尝试将监测范围扩大到严重程度、发病情况和护理来源各异的其他医学博士。我们的回顾性监测包括在研究期间（2007 年 1 月至 2011 年 12 月）之前或期间（2007 年 1 月至 2011 年 12 月）被诊断患有九种符合资格的 MD 之一的个人、一次或多次健康遭遇以及研究期间任何时间居住在美国四个地点之一（亚利桑那州、科罗拉多州、爱荷华州或纽约州西部）的个人。我们开发了用于病历提取、临床审查和数据池的病例定义、监测方案和软件应用程序。潜在病例根据国际疾病分类第九版临床修订版 (ICD-9-CM) 代码 359.0、359.1 和 359.21 以及国际疾病分类第十版修订版 (ICD-10) 代码 G71.0 和 G71.1 进行识别。按 MD 类型比较描述性统计数据。计算了每个 ICD-9-CM 代码识别的 MD 病例的百分比。结果 结论 2,862 例患者中，32.9% 为肌强直，25.8% 为 DBMD 营养不良，9.7% 为面肩肱 MD，9.1% 为肢带 MD。大多数病例是男性（63.6%）、非西班牙裔（59.8%）和白人（80.2%）。大约一半的病例是在自身（39.1%）或家人（6.2%）中进行基因诊断的。大约一半有MD家族史（48.9%）。遗传性渐进性 MD 代码 (359.1) 是识别符合条件的病例的最常见代码。强直性肌营养不良代码 (359.21) 识别出 83.4% 符合条件的强直性肌营养不良病例 (786/943)。 MD STARnet 是美国唯一适用于 MD 的多站点、基于人群的主动监测系统。继续扩大监测范围将提供有关多名医学博士的重要流行病学和健康结果信息。
Background Methods 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. 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 Conclusions 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). 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.