在有 20 年蛋白尿病史的三胞胎中鉴定 CUBN 变体。

IF 1 Q4 UROLOGY & NEPHROLOGY
Natsumi Yamamura-Miyazaki, Nana Sakakibara, Kandai Nozu, Yuko Shima, Kenichi Satomura, Satoko Yamamoto, Minato Baba, Kaori Fujiwara, Katsusuke Yamamoto, Toshimi Michigami
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引用次数: 0

摘要

CUBN 编码立方体蛋白,它在近端肾小管重吸收肾小球滤过的白蛋白中发挥作用。与 CUBN 相关的蛋白尿最近被确定为一种新的疾病概念,可能存在于以前在遗传学或组织学上未确诊的蛋白尿病例中。我们在此报告了一例三胞胎病例,他们在发病 20 年后被诊断出患有 CUBN 变体引起的慢性良性蛋白尿。原告是三胞胎中的第一个孩子,在新生儿期曾多次检测出尿蛋白阳性。3 岁时的尿液筛查也呈阳性。蛋白尿在非肾病范围内持续多年。8 岁时进行的肾活检发现肾小球有轻微异常。开始使用肾素-血管紧张素系统抑制剂治疗白蛋白性蛋白尿,但效果不佳。由于另外两个三胞胎的病程相似,因此对 NPHS1/2 和 WT1 基因进行了分析,但未发现异常。三胞胎在 15 岁时转为成人护理。2020 年,CUBN 相关蛋白尿被报道;因此,我们重新分析了他们的 DNA 样本,并在所有三胞胎中发现了 CUBN 复合杂合变异。CUBN相关蛋白尿的分子诊断将使患者免于不必要的治疗和对肾脏预后的担忧。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Identification of CUBN variants in triplets with a 20-year history of proteinuria.

CUBN encodes cubilin, which plays a role in the reabsorption of glomerular-filtered albumin in the proximal tubule. CUBN-related proteinuria was recently established as a new disease concept and may be present in proteinuric cases that were previously undiagnosed either genetically or histologically. We herein report a case of triplets diagnosed with chronic benign proteinuria due to CUBN variants 20 years after its onset. The proband, the first child of triplets, tested positive for urinary protein several times during the neonatal period. A urine screening test at 3 years old was positive. Proteinuria persisted for years within a non-nephrotic range. Kidney biopsy at 8 years old revealed minor glomerular abnormalities. Renin-angiotensin system inhibitors were started for albumin-based proteinuria but were ineffective. Since the two other triplets had similar courses, analyses of the NPHS1/2 and WT1 genes were performed but revealed no abnormalities. The triplets transitioned to adult care at 15 years old. CUBN-related proteinuria was reported in 2020; therefore, we re-analyzed their DNA samples and identified compound heterozygous variants in CUBN in all three triplets. The molecular diagnosis of CUBN-related proteinuria will save patients from unnecessary treatments and concerns about renal prognosis.

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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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