Literature DB >> 12478350

Phenotypic and genotypic features of Alport syndrome in Chinese children.

Fang Wang1, Jie Ding, Shunhua Guo, Jiyun Yang.   

Abstract

Chinese Alport syndrome (AS) was analyzed in 44 unrelated patients who were screened for mutations in the COL4A5 gene by polymerase chain reaction (PCR)-single-strand conformation polymorphism analysis or PCR direct sequencing in 30 of the 44 patients. The clinical data showed that all patients had hematuria; 25 of 29 male patients (86%) and 9 of 15 female patients (60%) had proteinuria; 11 of 29 male patients (38%) and 1 of 15 female patients (7%) had nephrotic-level proteinuria; 10 of 21 male patients examined (48%) and 1 of 12 female patients examined (8%) had hearing abnormalities. Renal function remained normal despite hearing abnormalities, and ocular lesions occurred in 10%. Among 30 of 44 patients who had a family history of end-stage renal disease (ESRD), 80% (24/30) belonged to X-linked juvenile kindreds, and 20% (6/30) patients to adult kindreds. Of the 44 patients, 14 did not have a family history of ESRD, while 11 of 14 patients diagnosed with X-linked AS did. DNA analysis revealed four missense mutations, two silent mutations, one substitution, and one in-frame deletion. PCR along with Southern hybridization analysis revealed a large deletion of the paired COL4A5 and COL4A6 genes. Chinese AS patients were characterized clinically with hematuria, heavy proteinuria, and more juvenile forms. Mutations in these patients were usually small mutations, while a large deletion involving the 5' part of both COL4A5 and COL4A6 genes was identified.

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Year:  2002        PMID: 12478350     DOI: 10.1007/s00467-002-0972-5

Source DB:  PubMed          Journal:  Pediatr Nephrol        ISSN: 0931-041X            Impact factor:   3.714


  5 in total

1.  Genotype-phenotype correlation in X-linked Alport syndrome.

Authors:  Mir Reza Bekheirnia; Berenice Reed; Martin C Gregory; Kim McFann; Alireza Abdollah Shamshirsaz; Amirali Masoumi; Robert W Schrier
Journal:  J Am Soc Nephrol       Date:  2010-04-08       Impact factor: 10.121

2.  A novel mutation of NPHS2 identified in a Chinese family.

Authors:  Zihua Yu; Jie Ding; Na Guan; Yan Shi; Jingjing Zhang; Jianping Huang; Yong Yao; Jiyun Yang
Journal:  Pediatr Nephrol       Date:  2004-11       Impact factor: 3.714

3.  Effect of heterozygous pathogenic COL4A3 or COL4A4 variants on patients with X-linked Alport syndrome.

Authors:  Yanqin Zhang; Jie Ding; Hongwen Zhang; Yong Yao; Huijie Xiao; Suxia Wang; Fang Wang
Journal:  Mol Genet Genomic Med       Date:  2019-03-18       Impact factor: 2.183

4.  Reassessing the pathogenicity of c.2858G>T(p.(G953V)) in COL4A5 Gene: report of 19 Chinese families.

Authors:  Yanqin Zhang; Jie Ding; Suxia Wang; Hongwen Zhang; Xuhui Zhong; Xiaoyu Liu; Ke Xu; Fang Wang
Journal:  Eur J Hum Genet       Date:  2019-10-01       Impact factor: 4.246

Review 5.  Novel Mutations of COL4A5 Identified in Chinese Families with X-Linked Alport Syndrome and Literature Review.

Authors:  Wen-Yu Gong; Fan-Na Liu; Liang-Hong Yin; Jun Zhang
Journal:  Biomed Res Int       Date:  2021-03-02       Impact factor: 3.411

  5 in total

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