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加强并利用主要研究者与患者权益倡导组织在罕见病临床研究中的合作——罕见病临床研究网络会议报告

Enhancing and leveraging principal investigator and patient advocacy group collaboration in rare disease clinical research-meeting report from the rare Diseases Clinical Research Network.

作者信息

Wheeden Kristen, Meyers Sheridan, Anthony Kristin, Chehade Mirna, Gifford Rogerwene C, King Eileen C, Seid Michael

机构信息

United Porphyrias Association, 4800 Hampden Lane, Suite 200, Bethesda, MD 20814, USA.

Cincinnati Children's Hospital Medical Center, Cincinatti, OH, USA.

出版信息

Ther Adv Rare Dis. 2025 Jul 11;6:26330040251357318. doi: 10.1177/26330040251357318. eCollection 2025 Jan-Dec.

Abstract

The Rare Diseases Clinical Research Network (RDCRN) works toward faster diagnosis and better treatment for people living with rare diseases, specifically by advancing clinical trial readiness. Inclusion of patient advocacy groups (PAGs) is mandated for each RDCRN consortia; principal investigator (PI)-PAG collaboration is expected to accelerate clinical trial readiness. Real-world examples of PI-PAG collaboration in rare disease clinical research (RDCR) are often not documented nor shared. We report on the Spring 2023 RDCRN meeting, which was dedicated to (a) capturing examples of ways that PAGs and PIs in the RDCRN collaborate, and (b) describing challenges and potential best practices for PAG-PI collaboration. PI and PAG attendees included 50 investigators and staff from 19 consortia and 41 PAG members from 21 consortia. Examples of collaboration in Study Design, Planning and Execution, Funding, and Stakeholder Engagement were captured, as were best practices and challenges to PI-PAG collaboration. Strengthening PI-PAG collaboration can accelerate rare disease research. Documenting real-world examples, and barriers and facilitators of collaboration, from across the RDCRN, supports existing frameworks for accelerating clinical trial readiness.

摘要

罕见病临床研究网络(RDCRN)致力于为罕见病患者实现更快的诊断和更好的治疗,特别是通过提高临床试验的准备程度来实现这一目标。每个RDCRN联盟都被要求纳入患者权益倡导组织(PAGs);主要研究者(PI)与PAG的合作有望加快临床试验的准备进程。在罕见病临床研究(RDCR)中,PI与PAG合作的实际案例往往没有记录或分享。我们报告了2023年春季RDCRN会议,该会议致力于(a)收集RDCRN中PAG与PI合作方式的案例,以及(b)描述PAG与PI合作面临的挑战和潜在的最佳实践。PI和PAG的参会人员包括来自19个联盟的50名研究人员和工作人员,以及来自21个联盟的41名PAG成员。会议收集了在研究设计、规划与执行、资金筹集以及利益相关者参与等方面的合作案例,以及PI与PAG合作的最佳实践和挑战。加强PI与PAG的合作可以加速罕见病研究。记录来自整个RDCRN的实际案例以及合作的障碍与促进因素,有助于支持现有的提高临床试验准备程度的框架。

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