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常染色体隐性多囊肾病的ARegPKD注册研究的最新进展

Recent Progress of the ARegPKD Registry Study on Autosomal Recessive Polycystic Kidney Disease.

作者信息

Ebner Kathrin, Schaefer Franz, Liebau Max Christoph

机构信息

Department of Pediatrics, University Hospital of Cologne , Cologne , Germany.

Division of Pediatric Nephrology, Centre for Pediatrics and Adolescent Medicine, Heidelberg University Medical Centre , Heidelberg , Germany.

出版信息

Front Pediatr. 2017 Feb 16;5:18. doi: 10.3389/fped.2017.00018. eCollection 2017.

Abstract

Autosomal recessive polycystic kidney disease (ARPKD) is a rare monogenic disease with a severe phenotype often presenting prenatally or in early childhood. With its obligate renal and hepatic involvement, ARPKD is one of the most important indications for liver and/or kidney transplantation in childhood. Marked phenotypic variability is observed, the genetic basis of which is largely unknown. Treatment is symptomatic and largely empiric as evidence-based guidelines are lacking. Therapeutic initiatives for ARPKD face the problem of highly variable cohorts and lack of clinical or biochemical risk markers without clear-cut clinical end points. ARegPKD is an international, multicenter, retro- and prospective, observational study to deeply phenotype patients with the clinical diagnosis of ARPKD. Initiated in 2013 as a web-based registry (www.aregpkd.org), ARegPKD enrolls patients across large parts of Europe and neighboring countries. By January 2017, more than 400 patients from 17 mostly European countries have been registered in the ARPKD registry study with significant follow-up data. Due to comprehensive retro- and prospective data collection and associated biobanking, ARegPKD will generate a unique ARPKD cohort with detailed longitudinal clinical characterization providing a basis for future clinical trials as well as translational research. Hence, ARegPKD is hoped to contribute to the pathophysiological understanding of the disease and to the improvement of clinical management.

摘要

常染色体隐性多囊肾病(ARPKD)是一种罕见的单基因疾病,具有严重的表型,常在产前或幼儿期出现。由于其必然累及肾脏和肝脏,ARPKD是儿童肝和/或肾移植的最重要指征之一。观察到明显的表型变异性,但其遗传基础大多未知。由于缺乏循证指南,治疗以对症为主且很大程度上是经验性的。ARPKD的治疗举措面临着队列高度异质性以及缺乏无明确临床终点的临床或生化风险标志物的问题。ARegPKD是一项国际多中心回顾性和前瞻性观察性研究,旨在深入了解临床诊断为ARPKD的患者的表型。ARegPKD于2013年作为基于网络的注册研究(www.aregpkd.org)启动,招募欧洲大部分地区及周边国家的患者。截至2017年1月,来自17个主要欧洲国家的400多名患者已在ARPKD注册研究中登记,并拥有大量随访数据。由于全面的回顾性和前瞻性数据收集以及相关的生物样本库建设,ARegPKD将生成一个独特的ARPKD队列,具有详细的纵向临床特征,为未来的临床试验以及转化研究提供基础。因此,希望ARegPKD有助于对该疾病的病理生理理解并改善临床管理。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f9c7/5327862/55ce32868460/fped-05-00018-g001.jpg

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