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肾脏囊性疾病:从实验台到病床边

Cystic Diseases of the Kidneys: From Bench to Bedside.

作者信息

Raina Rupesh, Lomanta Francis, Singh Siddhartha, Anand Alisha, Kalra Riti, Enukonda Vignasiddh, Barat Oren, Pandher Davinder, Sethi Sidharth K

机构信息

Department of Pediatric Nephrology, Akron Children's Hospital, Akron, Ohio, USA.

Department of Nephrology, Akron Nephrology Associates/Cleveland Clinic Akron General Medical Center, Akron, USA.

出版信息

Indian J Nephrol. 2023 Mar-Apr;33(2):83-92. doi: 10.4103/ijn.ijn_318_21. Epub 2023 Feb 20.

Abstract

Exploration into the causes of hereditary renal cystic diseases demonstrates a deep-rooted connection with the proteomic components of the cellular organelle cilia. Cilia are essential to the signaling cascades, and their dysfunction has been tied to a range of renal cystic diseases initiating with studies on the oak ridge polycystic kidney (ORPK) mouse model. Here, we delve into renal cystic pathologies that have been tied with ciliary proteosome and highlight the genetics associated with each. The pathologies are grouped based on the mode of inheritance, where inherited causes that result in cystic kidney disease phenotypes include autosomal dominant and autosomal recessive polycystic kidney disease, nephronophthisis (Bardet-Biedl syndrome and Joubert Syndrome), and autosomal dominant tubulointerstitial kidney disease. Alternatively, phakomatoses-, also known as neurocutaneous syndromes, associated cystic kidney diseases include tuberous sclerosis (TS) and Von Hippel-Lindau (VHL) disease. Additionally, we group the pathologies by the mode of inheritance to discuss variations in recommendations for genetic testing for biological relatives of a diagnosed individual.

摘要

对遗传性肾囊性疾病病因的探索表明,其与细胞器纤毛的蛋白质组学成分有着根深蒂固的联系。纤毛对于信号级联反应至关重要,自对橡树岭多囊肾(ORPK)小鼠模型的研究开始,其功能障碍就与一系列肾囊性疾病相关联。在此,我们深入探讨与纤毛蛋白酶体相关的肾囊性病变,并强调与之相关的遗传学特征。这些病变根据遗传模式进行分类,导致肾囊性疾病表型的遗传病因包括常染色体显性和常染色体隐性多囊肾病、肾单位肾痨(巴德-比埃尔综合征和儒贝尔综合征)以及常染色体显性肾小管间质性肾病。另外,错构瘤病(也称为神经皮肤综合征)相关的肾囊性疾病包括结节性硬化症(TS)和冯·希佩尔-林道(VHL)病。此外,我们根据遗传模式对这些病变进行分类,以讨论针对已确诊个体的生物学亲属进行基因检测的建议差异。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f1a3/10208543/60d6ae2d443c/IJN-33-83-g001.jpg

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