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国际纤维性骨发育不良进展性(FOP)协会全球注册中心的自我报告的基线表型。

Self-reported baseline phenotypes from the International Fibrodysplasia Ossificans Progressiva (FOP) Association Global Registry.

机构信息

Department of Medicine, Mayo Clinic School of Medicine, Mayo Clinic, Rochester, MN, United States.

BioSAS Consulting, Inc., Wellesley, MA, United States.

出版信息

Bone. 2020 May;134:115274. doi: 10.1016/j.bone.2020.115274. Epub 2020 Feb 13.

Abstract

A global, patient-reported registry has been established to characterize the course of disease and track clinical outcomes in patients with fibrodysplasia ossificans progressiva (FOP), an ultra-rare genetic condition of progressive heterotopic ossification (HO) that results in ankylosis of joints and renders most affected individuals immobile by the second decade of life. Here, we present baseline phenotypes on 299 patients (median age 21 years; range 0.1 to 78 years) from 54 countries based on aggregate data from the International FOP Association (IFOPA) Global Registry (the "FOP Registry"). The mean current age of the patients is 23.7 years (range, 0.1 to 78 years). Baseline characteristics are presented for FOP diagnosis, HO, flare-ups and precedent events, system-based prevalent symptomatology, encounters with medical and dental care providers, Patient Reported Outcomes Measurement Information System (PROMIS) Global Health Scale scores, physical function, as well as the use of aids, assistive devices, and adaptations. Correlations of PROMIS Global Health scores with HO burden and physical function are calculated. Associations of joint mobility with PROMIS Global Health scores, physical function, and use of aids, assistive devices, and adaptations are summarized. Overall, the FOP Registry database contains a broad sample of the global FOP patient population, providing a useful tool for expanding knowledge of FOP, designing clinical trials and facilitating evidence-based decisions about the optimal monitoring and management of affected individuals.

摘要

一个全球性的、基于患者报告的注册系统已经建立,旨在描述成骨不全性纤维发育不良(FOP)患者的疾病进程,并跟踪其临床结局。FOP 是一种罕见的遗传性疾病,其特征为进行性异位骨化(HO),导致关节强直,大多数受影响个体在生命的第二个十年就丧失活动能力。在此,我们根据国际 FOP 协会(IFOPA)全球注册中心(“FOP 注册中心”)的汇总数据,介绍了来自 54 个国家的 299 名患者(中位年龄 21 岁;年龄范围 0.1 至 78 岁)的基线表型。这些患者的平均当前年龄为 23.7 岁(年龄范围 0.1 至 78 岁)。介绍了 FOP 诊断、HO、发作和前期事件、基于系统的普遍症状、与医疗和牙科保健提供者的接触、患者报告的测量信息系统(PROMIS)全球健康量表评分、身体功能以及辅助器具、辅助设备和适应的使用情况等方面的基线特征。计算了 PROMIS 全球健康评分与 HO 负担和身体功能的相关性。总结了关节活动度与 PROMIS 全球健康评分、身体功能以及辅助器具、辅助设备和适应的使用之间的关联。总体而言,FOP 注册中心数据库包含了全球 FOP 患者群体的广泛样本,为扩展对 FOP 的认识、设计临床试验以及促进有关受影响个体的最佳监测和管理的循证决策提供了有用的工具。

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