A monocenter study on pediatric Alport syndrome featuring nephrotic syndrome as the primary manifestation

Author:

Qian Pei1ORCID

Affiliation:

1. Xi'an Jiaotong University

Abstract

Abstract

Background: Alport Syndrome (AS) is a genetic kidney disorder characterized by progressive kidney failure. It is often misdiagnosed as other kidney diseases due to its clinical phenotypic heterogeneity and the lack of specific clinical symptoms in early childhood. Methods: This study retrospectively analyzed clinical data of 7 pediatric patients admitted to Xi'an Children's Hospital between 2016 and 2022 due to clinical manifestations of nephrotic syndrome. Results: The 7 patients were from six families, and 4 patients had a family history of kidney disease. The median(IQR) age at presentation was 9.8 (7.8, 10.8) years and median follow-up was 4.4 (2.4–8.0) years.They all had hematuria, nephrotic proteinuria and hypoproteinemia. Kidney biopsy revealed focal segmental glomerulosclerosis (FSGS) on light microscopy. Among the patients, 8 pathogenic gene mutations were detected, 6 patients had mutations in the COL4A5gene. Furthermore, the mutations in 6 patients (85.7%) were severe.Treatment involved administering renin-angiotensin-aldosterone system (RAAS) inhibitors to all the patients starting from their first visit. Up to the present follow-up time, all the 7 patients exhibited varying degrees of reduction in proteinuria, with 1 of them experiencing kidney function decline, and 1 progressing to end-stage kidney disease (ESKD). Conclusion: AS should be considered in patients co-exhibiting nephrotic syndrome and hematuria, especially those with a poor response to steroid therapy or with a family history of hematuria. Additionally, AS should be considered in the genetic diagnosis of patients with kidney pathology consistent with FSGS. The most common pathogenic gene in AS patients with nephrotic syndrome is the COL4A5 gene, and most of them have severe mutations.

Publisher

Research Square Platform LLC

同舟云学术

1.学者识别学者识别

2.学术分析学术分析

3.人才评估人才评估

"同舟云学术"是以全球学者为主线,采集、加工和组织学术论文而形成的新型学术文献查询和分析系统,可以对全球学者进行文献检索和人才价值评估。用户可以通过关注某些学科领域的顶尖人物而持续追踪该领域的学科进展和研究前沿。经过近期的数据扩容,当前同舟云学术共收录了国内外主流学术期刊6万余种,收集的期刊论文及会议论文总量共计约1.5亿篇,并以每天添加12000余篇中外论文的速度递增。我们也可以为用户提供个性化、定制化的学者数据。欢迎来电咨询!咨询电话:010-8811{复制后删除}0370

www.globalauthorid.com

TOP

Copyright © 2019-2024 北京同舟云网络信息技术有限公司
京公网安备11010802033243号  京ICP备18003416号-3