IF 1 Q4 UROLOGY & NEPHROLOGY
Takamitsu Shiiya, Hirofumi Watanabe, Ryo Aida, Tadashi Otsuka, Ryohei Kaseda, Suguru Yamamoto, Yoshikatsu Kaneko, Shin Goto, Ichiei Narita
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引用次数: 0

摘要

常染色体显性多囊肾(ADPKD)是最常见的单基因肾脏疾病之一。诊断 ADPKD 需要影像学检查发现多发性肾囊肿,或在家族史不明的情况下进行基因检测。我们报告了一名因发现罕见的 PKD1 变异而被诊断为 ADPKD 的患者。患者是一名41岁的女性,偶然发现其肾脏有囊肿和钙化。全外显子组测序发现了一个罕见的PKD1变异体(NM_001009944.3:c.11557G > A/p.E3853K)。PKD1-PKD2 复合物的蛋白质结构预测显示,该变异体可能是致病的,因此诊断为常染色体显性多囊肾病。详细的家族病史显示,她的亲属也患有 ADPKD,这进一步支持了 ADPKD 的诊断。综合遗传分析和蛋白质结构预测导致了 ADPKD 的诊断,并确定了罕见的致病基因。这些方法有助于诊断病因不明的遗传性肾脏疾病。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Genetic and protein structure prediction analyses identify a rare pathogenic PKD1 variant causing autosomal dominant polycystic kidney disease.

Autosomal dominant polycystic kidney disease (ADPKD) is one of the most common monogenic kidney disorders. The diagnosis of ADPKD requires imaging findings showing multiple kidney cysts or genetic testing, in cases where a family history is unknown. We report a patient diagnosed with ADPKD based on the identification of a rare PKD1 variant. The patient was a 41-year-old female in whom cysts and calcification in the kidneys were incidentally detected. Whole-exome sequencing identified a rare PKD1 variant (NM_001009944.3: c.11557G > A/p.E3853K). Protein structure prediction of the PKD1-PKD2 complex showed that the variant may be pathogenic, leading to the diagnosis of autosomal dominant polycystic kidney disease. A detailed family history revealed that her relatives also had ADPKD, further supporting the diagnosis of ADPKD. Comprehensive genetic analysis and protein structure prediction led to the diagnosis of ADPKD and the identification of rare causative genes. These methods are useful for diagnosing hereditary kidney diseases of unknown etiologies.

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来源期刊
CEN Case Reports
CEN Case Reports UROLOGY & NEPHROLOGY-
CiteScore
1.90
自引率
0.00%
发文量
80
期刊介绍: Clinical and Experimental Nephrology (CEN) Case Reports is a peer-reviewed online-only journal, officially published biannually by the Japanese Society of Nephrology (JSN).  The journal publishes original case reports in nephrology and related areas.  The purpose of CEN Case Reports is to provide clinicians and researchers with a forum in which to disseminate their personal experience to a wide readership and to review interesting cases encountered by colleagues all over the world, from whom contributions are welcomed.
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