COQ8B基因缺乏是儿童肾移植受者视网膜异常的潜在原因。

IF 3.2 2区 生物学 Q3 BIOCHEMISTRY & MOLECULAR BIOLOGY
Yonghua Feng, Yi Feng, Zhigang Wang, Wenjing Li, Haowei Zhu, Zhou Li, Chenghao Feng, Hongen Xu, Guiwen Feng, Di Zhang, Wenjun Shang
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引用次数: 0

摘要

COQ8B基因缺乏引起的辅酶Q10合成障碍是儿童终末期肾病(ESRD)最常见的原因之一,通常表现为孤立性肾脏疾病,零星病例与肾外症状相关,如视网膜色素变性(RP)。通过对我中心26例COQ8B变异肾移植患儿的长期随访发现,尽管肾移植结果良好,但仍有23.1%的患儿出现夜盲症或其他眼部症状。我们招募了9名儿童进行系统的眼科检查,发现其中3名儿童明确诊断为RP,其余儿童均有不同程度的视网膜异常,无论是否感到眼部不适。与未携带该基因变异的肾移植患儿相比,携带COQ8B基因变异患儿的ERG结果明显较差,这是早期诊断RP的重要指标。在筛查这些儿童的WES数据中未发现已知的rp相关致病基因突变。转录组分析表明,COQ8B基因突变与RP之间的潜在关联与ATP合成、氧化磷酸化和光转导途径有关。这项研究首次提出,COQ8B基因缺乏导致肾移植儿童视网膜疾病不是散发性的。补充辅酶Q10可能对COQ8B突变儿童肾移植后视网膜有保护作用,需要进一步研究和临床关注。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
COQ8B gene deficiency as a potential cause of retinal abnormalities in Pediatric kidney transplant recipients.

Coenzyme Q10 synthesis disorder caused by COQ8B gene deficiency is among the most prevalent causes of end-stage renal disease (ESRD) in children, which usually presents as isolated kidney disease, with sporadic cases associated with extrarenal symptoms such as retinitis pigmentosa (RP). Through long-term follow-up of 26 renal transplant children with COQ8B variants at our center, it is observed that, despite favorable renal transplant outcomes, 23.1% of children experienced night blindness or other ocular symptoms. Nine children were recruited for systematic ophthalmic examination and found that three of them were definitely diagnosed with RP, and the remaining children had varying degrees of retinal abnormalities regardless of perceived ocular discomfort. Compared with kidney transplant children without this genetic variant, children with the COQ8B genetic variant had significantly worse ERG results, which was an important indicator for the early diagnosis of RP. No known RP-associated pathogenic gene mutations were identified in the WES data of these children upon screening. Transcriptome analysis suggested that the potential association between COQ8B gene mutations and RP was related to ATP synthesis, oxidative phosphorylation, and phototransduction pathways. This study was the first to propose that COQ8B gene deficiency leading to retinal disorders in kidney transplanted children is not sporadic. Coenzyme Q10 supplementation may have a protective effect on the retina after renal transplantation in children with COQ8B mutations, requiring further research and clinical attention.

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来源期刊
Human molecular genetics
Human molecular genetics 生物-生化与分子生物学
CiteScore
6.90
自引率
2.90%
发文量
294
审稿时长
2-4 weeks
期刊介绍: Human Molecular Genetics concentrates on full-length research papers covering a wide range of topics in all aspects of human molecular genetics. These include: the molecular basis of human genetic disease developmental genetics cancer genetics neurogenetics chromosome and genome structure and function therapy of genetic disease stem cells in human genetic disease and therapy, including the application of iPS cells genome-wide association studies mouse and other models of human diseases functional genomics computational genomics In addition, the journal also publishes research on other model systems for the analysis of genes, especially when there is an obvious relevance to human genetics.
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