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遗传性肾病综合征中基因研究结果的转化:缺失的环节。

Translating genetic findings in hereditary nephrotic syndrome: the missing loops.

作者信息

Hall Gentzon, Gbadegesin Rasheed A

机构信息

Department of Medicine and Duke Molecular Physiology Institute, Duke University Medical Center, Durham, North Carolina; and.

Department of Pediatrics and Duke Molecular Physiology Institute, Duke University Medical Center, Durham, North Carolina

出版信息

Am J Physiol Renal Physiol. 2015 Jul 1;309(1):F24-8. doi: 10.1152/ajprenal.00683.2014. Epub 2015 Mar 25.

Abstract

Nephrotic syndrome (NS) is a clinicopathological entity characterized by proteinuria, hypoalbuminemia, peripheral edema, and hyperlipidemia. It is the most common cause of glomerular disease in children and adults. Although the molecular pathogenesis of NS is not completely understood, data from the study of familial NS suggest that it is a "podocytopathy." Virtually all of the genes mutated in hereditary NS localize to the podocyte or its secreted products and the slit diaphragm. Since the completion of human genome sequence and the advent of next generation sequencing, at least 29 causes of single-gene NS have been identified. However, these findings have not been matched by therapeutic advances owing to suboptimal in vitro and in vivo models for the study of human glomerular disease and podocyte injury phenotypes. Multidisciplinary collaboration between clinicians, geneticists, cell biologists, and molecular physiologists has the potential to overcome this barrier and thereby speed up the translation of genetic findings into improved patient care.

摘要

肾病综合征(NS)是一种临床病理实体,其特征为蛋白尿、低白蛋白血症、外周水肿和高脂血症。它是儿童和成人肾小球疾病最常见的病因。尽管NS的分子发病机制尚未完全明确,但家族性NS的研究数据表明它是一种“足细胞病”。实际上,遗传性NS中发生突变的所有基因都定位于足细胞或其分泌产物以及裂孔隔膜。自人类基因组序列完成和下一代测序技术出现以来,已鉴定出至少29种单基因NS的病因。然而,由于用于研究人类肾小球疾病和足细胞损伤表型的体外和体内模型欠佳,这些发现尚未带来治疗上的进展。临床医生、遗传学家、细胞生物学家和分子生理学家之间的多学科合作有潜力克服这一障碍,从而加速将遗传学发现转化为改善患者护理的进程。

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