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多囊肾病分子诊断的最新进展。

Recent advances in the molecular diagnosis of polycystic kidney disease.

机构信息

a Center for Human Genetics , Bioscientia , Ingelheim , Germany.

b Department of Medicine , University Hospital Freiburg , Freiburg , Germany.

出版信息

Expert Rev Mol Diagn. 2017 Dec;17(12):1037-1054. doi: 10.1080/14737159.2017.1386099. Epub 2017 Nov 16.

Abstract

Polycystic kidney disease (PKD) is clinically and genetically heterogeneous and constitutes the most common heritable kidney disease. Most patients are affected by the autosomal dominant form (ADPKD) which generally is an adult-onset multisystem disorder. By contrast, the rarer recessive form ARPKD usually already manifests perinatally or in childhood. In some patients, however, ADPKD and ARPKD can phenotypically overlap with early manifestation in ADPKD and only late onset in ARPKD. Progressive fibrocystic renal changes are often accompanied by severe hepatobiliary changes or other extrarenal abnormalities. Areas covered: A reduced dosage of disease proteins disturbs cell homeostasis and explains a more severe clinical course in some PKD patients. Cystic kidney disease is also a common feature of other ciliopathies and genetic syndromes. Genetic diagnosis may guide clinical management and helps to avoid invasive measures and to detect renal and extrarenal comorbidities early in the clinical course. Expert Commentary: The broad phenotypic and genetic heterogeneity of cystic and polycystic kidney diseases make NGS a particularly powerful approach. Interpretation of data becomes the challenge and bench and bedside benefit from digitized multidisciplinary interrelationships.

摘要

多囊肾病 (PKD) 在临床上和遗传上具有异质性,是最常见的遗传性肾脏疾病。大多数患者受常染色体显性遗传形式 (ADPKD) 的影响,这种形式通常是一种成年发病的多系统疾病。相比之下,罕见的常染色体隐性遗传形式 ARPKD 通常在围产期或儿童期就已表现出来。然而,在一些患者中,ADPKD 和 ARPKD 可以在 ADPKD 中表现出早期表现,而在 ARPKD 中仅表现出晚期。进行性纤维囊性肾脏变化常伴有严重的肝胆变化或其他肾脏外异常。涵盖领域:疾病蛋白的低剂量表达会破坏细胞内稳态,并解释了一些 PKD 患者更严重的临床病程。囊性肾病也是其他纤毛病和遗传综合征的常见特征。基因诊断可以指导临床管理,有助于避免侵袭性措施,并在临床病程早期发现肾脏和肾脏外合并症。专家评论:囊性和多囊肾病的广泛表型和遗传异质性使得 NGS 成为一种特别强大的方法。数据解释成为了挑战,临床和床边受益于数字化多学科相互关系。

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