| Literature DB >> 31209800 |
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
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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