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在一个中国的 X 连锁 Alport 综合征家系中,对最初被诊断为 IgAN 的先证者进行鉴定,发现了一个新型的 COL4A5 突变。

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

机构信息

Department of Nephrology and Rheumatology, Hunan Children's Hospital, Academy of Pediatrics of University of South China, Changsha, 410007, China.

Shenzhen Seventh People's Hospital, Shenzhen, 518081, China.

出版信息

Sci China Life Sci. 2019 Dec;62(12):1572-1579. doi: 10.1007/s11427-018-9545-3. Epub 2019 Jun 17.

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.

摘要

阿尔波特综合征(AS)是一种遗传性进行性肾病,其特征为血尿、肾小球基底膜的超微结构病变、眼部病变和感觉神经性耳聋。COL4A5 种系突变与伴极端表型异质性的 X 连锁 AS 相关。在此,我们研究了一个有 AS 的中国家庭。先证者是一名 9 岁男孩,有血尿和蛋白尿。根据肾活检和免疫荧光的检测结果,先证者最初被诊断为 IgA 肾病,并建议相应治疗。同时,我们发现治疗效果不佳。因此,为了进行正确的临床诊断和适当的治疗,我们进行了基于外显子组的靶向下一代测序。我们在 COL4A5 基因中发现了一个新的杂合单核苷酸缺失 c.1902delA。家系分析表明,该新突变在受累家庭成员中共同遗传,但在未受累家庭成员中不存在。先证者的临床诊断修订为伴有 IgA 肾病的 AS,这很少有报道。我们的发现证明了遗传筛查应用的重要性,扩大了伴非典型肾脏表型的 COL4A5 相关 AS 患者的突变谱,并为我们在诊断过程中的曲折提供了一个很好的借鉴。

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