A practical framework to approach the development and evaluation of patient registries for rare diseases

构建和评估罕见病患者登记系统的实用框架

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Abstract

BACKGROUND: Patient registries are essential to rare disease research, but the implementation and maintenance of a registry requires substantial investments. Despite the large number of rare disease communities that currently have, or are planning for, a registry, there is little guidance on if and when a rare disease registry should be established. There is also little guidance on how to articulate registry objectives to address current and evolving scientific and community needs. MAIN BODY: A practical framework is needed to assist rare disease registry stakeholders to understand and prioritize registry objectives and requirements for registry design, data collection, and funding. In this review, we synthesize existing patient registry classifications and prior reports of rare disease registry experience into a unified framework characterizing rare disease registries by their types of research questions and scientific needs. CONCLUSION: This simple framework can be used to help rare disease communities identify their research requirements, and support decision making around whether to initiate, and how invest in, registry infrastructure.

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