常染色体显性阿尔波特综合征的难以捉摸的诊断:基因组测序是一个游戏规则改变者。

IF 1.3 Q3 MEDICINE, GENERAL & INTERNAL
Cureus Pub Date : 2025-10-07 eCollection Date: 2025-10-01 DOI:10.7759/cureus.94077
Achilleas Betsikos, Eleni Paschou, Virginia Geladari, Evanthia Gazouni, Nikolaos Sabanis
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引用次数: 0

摘要

我们报告一例55岁女性慢性无症状血尿,长期诊断为基底膜疾病,表现为高血压恶化和显著程度的蛋白尿进展为慢性肾脏疾病。基因测序鉴定出COL4A4基因(c.1321_1369+3del)的杂合致病性变异,这是一个52个碱基对的缺失,破坏了外显子20/内含子20连接的正常核糖核酸(RNA)剪接。In silico分析预测典型剪接位点的完全缺失,导致iv型胶原的剪接不当和α4链功能障碍。该家族的基因检测证实,在另外两代人中也存在相同的变异。据我们所知,这是第一个报道的家族中c.1321_1369+3del突变是常染色体显性Alport综合征的唯一原因。
本文章由计算机程序翻译,如有差异,请以英文原文为准。

An Elusive Diagnosis of Autosomal Dominant Alport Syndrome: Genomic Sequencing Is a Game Changer.

An Elusive Diagnosis of Autosomal Dominant Alport Syndrome: Genomic Sequencing Is a Game Changer.

An Elusive Diagnosis of Autosomal Dominant Alport Syndrome: Genomic Sequencing Is a Game Changer.

An Elusive Diagnosis of Autosomal Dominant Alport Syndrome: Genomic Sequencing Is a Game Changer.

We report the case of a 55-year-old woman with chronic asymptomatic hematuria and a longstanding diagnosis of thin basement membrane disease, presenting with worsening hypertension and a significant degree of proteinuria progressing to chronic kidney disease. Genetic sequencing identified a heterozygous pathogenic variant in the COL4A4 gene (c.1321_1369+3del), a 52-base pair deletion that disrupts normal ribonucleic acid (RNA) splicing in the exon 20/intron 20 junction. In silico analysis predicted the complete loss of the canonical splice site, leading to improper splicing and the dysfunction of the α4 chain of type-IV collagen. Genetic testing in the family confirmed the presence of the same variant in two additional generations. To our knowledge, this is the first reported family in which the c.1321_1369+3del mutation is the sole cause of autosomal dominant Alport syndrome.

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