{"title":"COQ8B基因缺乏是儿童肾移植受者视网膜异常的潜在原因。","authors":"Yonghua Feng, Yi Feng, Zhigang Wang, Wenjing Li, Haowei Zhu, Zhou Li, Chenghao Feng, Hongen Xu, Guiwen Feng, Di Zhang, Wenjun Shang","doi":"10.1093/hmg/ddaf084","DOIUrl":null,"url":null,"abstract":"<p><p>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.</p>","PeriodicalId":13070,"journal":{"name":"Human molecular genetics","volume":" ","pages":"1368-1379"},"PeriodicalIF":3.2000,"publicationDate":"2025-08-16","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":"{\"title\":\"COQ8B gene deficiency as a potential cause of retinal abnormalities in Pediatric kidney transplant recipients.\",\"authors\":\"Yonghua Feng, Yi Feng, Zhigang Wang, Wenjing Li, Haowei Zhu, Zhou Li, Chenghao Feng, Hongen Xu, Guiwen Feng, Di Zhang, Wenjun Shang\",\"doi\":\"10.1093/hmg/ddaf084\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>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.</p>\",\"PeriodicalId\":13070,\"journal\":{\"name\":\"Human molecular genetics\",\"volume\":\" \",\"pages\":\"1368-1379\"},\"PeriodicalIF\":3.2000,\"publicationDate\":\"2025-08-16\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Human molecular genetics\",\"FirstCategoryId\":\"99\",\"ListUrlMain\":\"https://doi.org/10.1093/hmg/ddaf084\",\"RegionNum\":2,\"RegionCategory\":\"生物学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"BIOCHEMISTRY & MOLECULAR BIOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Human molecular genetics","FirstCategoryId":"99","ListUrlMain":"https://doi.org/10.1093/hmg/ddaf084","RegionNum":2,"RegionCategory":"生物学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"BIOCHEMISTRY & MOLECULAR BIOLOGY","Score":null,"Total":0}
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.
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.