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Progress, challenges and global approaches to rare diseases.罕见病的进展、挑战与全球策略。
Acta Paediatr. 2021 Oct;110(10):2711-2716. doi: 10.1111/apa.15974. Epub 2021 Jun 19.
2
Capturing Data in Rare Disease Registries to Support Regulatory Decision Making: A Survey Study Among Industry and Other Stakeholders.罕见病登记处数据采集以支持监管决策制定:行业和其他利益相关者的调查研究。
Drug Saf. 2021 Aug;44(8):853-861. doi: 10.1007/s40264-021-01081-z. Epub 2021 Jun 6.
3
Lessons learned from hemolytic uremic syndrome registries: recommendations for implementation.从溶血尿毒综合征登记中吸取的经验教训:实施建议。
Orphanet J Rare Dis. 2021 May 25;16(1):240. doi: 10.1186/s13023-021-01871-9.
4
Supporting international networks through platforms for standardised data collection-the European Registries for Rare Endocrine Conditions (EuRRECa) model.通过标准化数据收集平台支持国际网络——欧洲罕见内分泌疾病注册中心(EuRRECa)模式。
Endocrine. 2021 Mar;71(3):555-560. doi: 10.1007/s12020-021-02617-0. Epub 2021 Jan 29.
5
The EuRRECa Project as a Model for Data Access and Governance Policies for Rare Disease Registries That Collect Clinical Outcomes.EuRRECa 项目作为罕见病登记处收集临床结果的数据访问和治理政策的典范。
Int J Environ Res Public Health. 2020 Nov 25;17(23):8743. doi: 10.3390/ijerph17238743.
6
The current landscape of European registries for rare endocrine conditions.欧洲罕见内分泌疾病登记处的现状。
Eur J Endocrinol. 2019 Jan 1;180(1):89-98. doi: 10.1530/EJE-18-0861.
7
Recommendations for Improving the Quality of Rare Disease Registries.改善罕见病登记处质量的建议。
Int J Environ Res Public Health. 2018 Aug 3;15(8):1644. doi: 10.3390/ijerph15081644.
8
A sustainable solution for the activities of the European network for surveillance of congenital anomalies: EUROCAT as part of the EU Platform on Rare Diseases Registration.欧洲先天性异常监测网络活动的可持续解决方案:作为欧盟罕见病注册平台一部分的EUROCAT
Eur J Med Genet. 2018 Sep;61(9):513-517. doi: 10.1016/j.ejmg.2018.03.008. Epub 2018 Mar 27.
9
Data Quality in Rare Diseases Registries.罕见病登记处的数据质量。
Adv Exp Med Biol. 2017;1031:149-164. doi: 10.1007/978-3-319-67144-4_8.
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An assessment of the quality of the I-DSD and the I-CAH registries - international registries for rare conditions affecting sex development.对I-DSD和I-CAH登记处质量的评估——影响性发育的罕见病症国际登记处。
Orphanet J Rare Dis. 2017 Mar 20;12(1):56. doi: 10.1186/s13023-017-0603-7.

罕见病登记处的质量评估——对疾病登记处基本特征的评估。

The Quality Evaluation of Rare Disease Registries-An Assessment of the Essential Features of a Disease Registry.

机构信息

Developmental Endocrinology Research Group, Royal Hospital for Children, University of Glasgow, Glasgow G51 4TF, UK.

Office for Rare Conditions, University of Glasgow, Glasgow G51 4TF, UK.

出版信息

Int J Environ Res Public Health. 2021 Nov 15;18(22):11968. doi: 10.3390/ijerph182211968.

DOI:10.3390/ijerph182211968
PMID:34831724
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8620980/
Abstract

Rare disease (RD) registries aim to promote data collection and sharing, and facilitate multidisciplinary collaboration with the overall aim of improving patient care. Recommendations relating to the minimum standards necessary to develop and maintain high quality registries are essential to ensure high quality data and sustainability of registries. The aim of this international study was to survey RD registry leaders to ascertain the level of consensus amongst the RD community regarding the quality criteria that should be considered essential features of a disease registry. Of 35 respondents representing 40 RD registries, over 95% indicated that essential quality criteria should include establishment of a good governance system (ethics approval, registry management team, standard operating protocol and long-term sustainability plan), data quality (personnel responsible for data entry and procedures for checking data quality) and construction of an IT infrastructure complying with Findable, Accessible, Interoperable and Reusable (FAIR) principles to maintain registries of high quality, with procedures for authorized user access, erasing personal data, data breach procedures and a web interface. Of the 22 registries that performed a self-assessment, over 80% stated that their registry had a leader, project management group, steering committee, active funding stream, website, and user access policies. This survey has acceptability amongst the RD community for the self-quality evaluation of RD registries with high levels of consensus for the proposed quality criteria.

摘要

罕见病(RD)登记处旨在促进数据收集和共享,并促进多学科合作,总体目标是改善患者护理。制定和维护高质量登记处所需的最低标准的建议对于确保高质量数据和登记处的可持续性至关重要。本国际研究旨在调查 RD 登记处负责人,以确定 RD 社区在应考虑作为疾病登记处基本特征的质量标准方面的共识程度。在代表 40 个 RD 登记处的 35 名受访者中,超过 95%的人表示,基本质量标准应包括建立良好的治理系统(伦理批准、登记处管理团队、标准操作协议和长期可持续性计划)、数据质量(负责数据录入的人员和检查数据质量的程序)和构建符合可发现性、可访问性、互操作性和可重用性(FAIR)原则的 IT 基础设施,以维护高质量的登记处,并有授权用户访问、删除个人数据、数据泄露程序和网络界面的程序。在进行自我评估的 22 个登记处中,超过 80%的登记处表示其有负责人、项目管理小组、指导委员会、积极的资金流、网站和用户访问政策。这项调查在 RD 社区中是可以接受的,因为它对 RD 登记处进行了自我质量评估,并且对拟议的质量标准达成了高度共识。