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遗传性结缔组织疾病:数据库描述和初步队列特征分析。

Heritable disorders of connective tissue: Description of a data repository and initial cohort characterization.

机构信息

Department of Medicine, The Pennsylvania State University, College of Medicine, Hershey, Pennsylvania.

Department of Surgery, The Pennsylvania State University, College of Medicine, Hershey, Pennsylvania.

出版信息

Am J Med Genet A. 2019 Apr;179(4):552-560. doi: 10.1002/ajmg.a.61054. Epub 2019 Feb 1.

Abstract

We describe a data repository on heritable disorders of connective tissue (HDCT) assembled by the National Institutes of Health's National Institute on Aging (NIA) Intramural Research Program between 2001 and 2013. Participants included affected persons with a wide range of heritable connective tissue phenotypes, and unaffected family members. Elements include comprehensive history and physical examination, standardized laboratory data, physiologic measures and imaging, standardized patient-reported outcome measures, and an extensive linked biorepository. The NIA made a commitment to make the repository available to extramural investigators and deposited samples at Coriell Tissue Repository (N = 126) and GenTAC registry (N = 132). The clinical dataset was transferred to Penn State University College of Medicine Clinical and Translational Science Institute in 2016, and data elements inventoried. The consented cohort of 1,009 participants averaged 39 ± 18 years (mean ± SD, range 2-95) at consent; gender distribution is 71% F and 83% self-report Caucasian ethnicity. Diagnostic categories include Ehlers-Danlos syndrome (classical N = 50, hypermobile N = 99, vascular N = 101, rare types and unclassified N = 178), Marfan syndrome (N = 33), Stickler syndrome (N = 60), fibromuscular dysplasia (N = 135), Other HDCT (N = 72). Unaffected family members (N = 218) contributed DNA for the molecular archive only. We aim to develop further discrete data from unstructured elements, analyze multisymptom HDCT manifestations, encourage data use by other researchers and thereby better understand the complexity of these high-morbidity conditions and their multifaceted effects on affected persons.

摘要

我们描述了一个由美国国立卫生研究院下属的国家老龄化研究所(NIA)于 2001 年至 2013 年期间收集的遗传性结缔组织疾病(HDCT)数据库。参与者包括具有广泛遗传性结缔组织表型的患者和未受影响的家庭成员。研究内容包括全面的病史和体格检查、标准化的实验室数据、生理学测量和影像学、标准化的患者报告结局测量以及广泛的相关生物样本库。NIA 承诺将该数据库提供给外部研究人员,并将样本存放在科里尔组织库(N=126)和 GenTAC 登记处(N=132)。临床数据集于 2016 年被转移到宾夕法尼亚州立大学医学院临床与转化科学研究所,并对数据元素进行了编目。在同意参与研究的 1009 名参与者中,平均年龄为 39±18 岁(平均值±标准差,范围 2-95);性别分布为 71%为女性,83%自报为白种人。诊断类别包括埃勒斯-当洛斯综合征(经典型 N=50、高活动型 N=99、血管型 N=101、罕见类型和未分类型 N=178)、马凡综合征(N=33)、斯蒂克勒综合征(N=60)、纤维肌性发育不良(N=135)、其他 HDCT(N=72)。未受影响的家庭成员(N=218)仅为分子档案提供了 DNA。我们旨在从非结构化元素中进一步开发离散数据,分析多症状 HDCT 表现,鼓励其他研究人员使用数据,从而更好地了解这些高发病率疾病的复杂性及其对患者的多方面影响。

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