一名 COQ2 肾小球病变患者的肾小球基底膜超微结构变化:病例报告。

IF 2.4 4区 医学 Q2 UROLOGY & NEPHROLOGY
Nephrology Pub Date : 2024-09-01 Epub Date: 2024-06-04 DOI:10.1111/nep.14329
Liuyu Sun, Yali Ren, Baige Su, Suxia Wang, Xuhui Zhong, Yuwu Jiang, Fang Wang
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引用次数: 0

摘要

由COQ2致病变体引起的原发性辅酶Q10缺乏症-1是一种常染色体隐性遗传疾病,基因检测是诊断这种疾病的金标准。本研究纳入了一名患有类固醇抵抗性肾病综合征、局灶节段性肾小球硬化症和进行性肾功能不全的中国男孩。电子显微镜检查发现,肾小球基底膜厚度不规则,呈片状,荚膜细胞中的足突弥漫性脱落,线粒体肿胀,荚膜细胞中的嵴异常。在出现轻度肾功能障碍约3周后开始补充辅酶Q10,但并未改善该患者的肾脏状况。原发性全外显子组测序和桑格测序发现了两个COQ2异源变异:第6外显子中的母系遗传新型变异c.1013G > A[p.(Gly338Glu)]和第7外显子中的未知来源变异c.1159C > T[p.(Arg387*)]。随后的长线程测序表明,这两个变异位于不同的等位基因上。我们的报告扩展了 COQ2 肾小球病的表型和基因型谱。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Glomerular basement membrane ultrastructural changes in a patient with COQ2 glomerulopathy: A case report.

Primary coenzyme Q10 deficiency-1, caused by COQ2 disease-causing variants, is an autosomal recessive disorder, and genetic testing is the gold standard for diagnosing this condition. A Chinese boy with steroid-resistant nephrotic syndrome, focal segmental glomerulosclerosis, and progressive kidney insufficiency was included in the study. Electron microscopy revealed the glomerular basement membrane with irregular thickness and lamellation with diffuse effacement of foot processes in the podocytes, and swollen mitochondria with abnormal cristae in the podocytes. Coenzyme Q10 supplementation started about 3 weeks after the onset of mild kidney dysfunction did not improve the proband's kidney outcome. Proband-only whole-exome sequencing and Sanger sequencing revealed two heteroallelic COQ2 variants: a maternally inherited novel variant c.1013G > A[p.(Gly338Glu)] in exon 6 and a variant of unknown origin c.1159C > T[p.(Arg387*)] in exon 7. Subsequent long-read sequencing demonstrated these two variants were located on different alleles. Our report extends the phenotypic and genotypic spectrum of COQ2 glomerulopathy.

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来源期刊
Nephrology
Nephrology 医学-泌尿学与肾脏学
CiteScore
4.50
自引率
4.00%
发文量
128
审稿时长
4-8 weeks
期刊介绍: Nephrology is published eight times per year by the Asian Pacific Society of Nephrology. It has a special emphasis on the needs of Clinical Nephrologists and those in developing countries. The journal publishes reviews and papers of international interest describing original research concerned with clinical and experimental aspects of nephrology.
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