Literature DB >> 31209800

Identification of a novel COL4A5 mutation in the proband initially diagnosed as IgAN from a Chinese family with X-linked Alport syndrome.

Zhihui Li1, Peng Zhu2, Hui Huang3, Ying Pan4, Peng Han3, Huanhuan Cui3, Zhijuan Kang4, Mai Xun4, Yi Zhang4, Saijun Liu3, Jian Wang5, Jing Wu6.   

Abstract

Alport syndrome (AS) is a hereditary progressive nephropathy characterized by hematuria, ultrastructural lesions of the glomerular basement membrane, ocular lesions and sensorineural hearing loss. Germline mutations of COL4A5 are associated with X-linked AS with an extreme phenotypic heterogeneity. Here, we investigated a Chinese family with Alport syndrome. The proband was a 9-year-old boy with hematuria and proteinuria. Based on the test results of renal biopsy and immunofluorescence, the proband was initially diagnosed as IgA nephropathy and the treatment was recommended accordingly. Meanwhile, we found that the treatment outcome was poor. Therefore, for proper clinical diagnosis and appropriate treatment, targeted exome-based next-generation sequencing has been undertaken. We identified a novel hemizygous single nucleotide deletion c.1902delA in COL4A5 gene. Segregation analysis identified that this novel mutation is co-segregated among the affected family members but absent in unaffected family members. The clinical diagnosis of the proband was revised as AS accompanied by IgA nephropathy, which has been rarely reported. Our findings demonstrated the significance of the application of Genetic screening, expanded the mutation spectrum of COL4A5 associated AS patients with atypical renal phenotypes and provided a good lesson to be learned from our detour during the diagnosis.

Entities:  

Keywords:  Alport syndrome; COL4A5; IgA nephropathy; a novel frameshift mutation; targeted exome-based next-generation sequencing

Mesh:

Substances:

Year:  2019        PMID: 31209800     DOI: 10.1007/s11427-018-9545-3

Source DB:  PubMed          Journal:  Sci China Life Sci        ISSN: 1674-7305            Impact factor:   6.038


  9 in total

1.  Preimplantation Genetic Testing Prevented Intergenerational Transmission of X-Linked Alport Syndrome.

Authors:  Xiaoling Hu; Jiahui Zhang; Yuan Lv; Xijing Chen; Guofang Feng; Liya Wang; Yinghui Ye; Fan Jin; Yimin Zhu
Journal:  Kidney Dis (Basel)       Date:  2021-09-09

2.  X-CNV: genome-wide prediction of the pathogenicity of copy number variations.

Authors:  Li Zhang; Jingru Shi; Jian Ouyang; Riquan Zhang; Yiran Tao; Dongsheng Yuan; Chengkai Lv; Ruiyuan Wang; Baitang Ning; Ruth Roberts; Weida Tong; Zhichao Liu; Tieliu Shi
Journal:  Genome Med       Date:  2021-08-18       Impact factor: 11.117

3.  Use of whole-exome sequencing to identify a novel ADCY10 mutation in a patient with nephrolithiasis.

Authors:  Chenyu Wang; Ran Du; Jieyuan Jin; Yi Dong; Jishi Liu; Liangling Fan; Rong Xiang
Journal:  Am J Transl Res       Date:  2020-08-15       Impact factor: 4.060

4.  An overview of the multi-pronged approach in the diagnosis of Alport syndrome for 22 children in Northeast China.

Authors:  Li Zhang; Bai-Chao Sun; Bing-Gang Zhao; Qing-Shan Ma
Journal:  BMC Nephrol       Date:  2020-07-23       Impact factor: 2.388

5.  Genotype-phenotype correlations of Berardinelli-Seip congenital lipodystrophy and novel candidate genes prediction.

Authors:  Meng Ren; Jingru Shi; Jinmeng Jia; Yongli Guo; Xin Ni; Tieliu Shi
Journal:  Orphanet J Rare Dis       Date:  2020-04-29       Impact factor: 4.123

6.  The First COL4A5 Exon 41A Glycine Substitution in a Family With Alport Syndrome.

Authors:  Fang Wang; Dan Zhao; Jie Ding; Xuejuan Li
Journal:  Front Pediatr       Date:  2020-04-09       Impact factor: 3.418

Review 7.  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

8.  Genetic Analysis of 28 Chinese Families With Tyrosinase-Positive Oculocutaneous Albinism.

Authors:  Linya Ma; Jianjian Zhu; Jing Wang; Yazhou Huang; Jibo Zhang; Chao Wang; Yuan Zhou; Dan Peng
Journal:  Front Genet       Date:  2021-10-11       Impact factor: 4.599

9.  A Novel Germline Compound Heterozygous Mutation of BRCA2 Gene Associated With Familial Peripheral Neuroblastic Tumors in Two Siblings.

Authors:  Yeran Yang; Jiwei Chen; Hong Qin; Yaqiong Jin; Li Zhang; Shen Yang; Huanmin Wang; Libing Fu; Enyu Hong; Yongbo Yu; Jie Lu; Yan Chang; Xin Ni; Min Xu; Tieliu Shi; Yongli Guo
Journal:  Front Genet       Date:  2021-07-23       Impact factor: 4.599

  9 in total

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