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Genotype–phenotype correlation of X-linked Alport syndrome observed in both genders: a multicenter study in South Korea

Title
Genotype–phenotype correlation of X-linked Alport syndrome observed in both genders: a multicenter study in South Korea
Authors
KimJi HyunLimSeon HeeSongJi YeonChoMyung HyunHyunHyeSunYangEun MiLeeJung WonMin HyunParkMin JiJoo HoonJungJiwonYooKee HwanJangKyung MiPaiKi SooSuhJin-SoonNamgoongMee KyungChungWoo YeongSu JinEun YoungKyung MinNam HeeMinsunPaikJin HoKangHee GyungAhnYo HanCheongHae Il
Ewha Authors
이정원
SCOPUS Author ID
이정원scopusscopus
Issue Date
2023
Journal Title
Scientific Reports
ISSN
2045-2322JCR Link
Citation
Scientific Reports vol. 13, no. 1
Publisher
Nature Research
Indexed
SCIE; SCOPUS WOS scopus
Document Type
Article
Abstract
The genotype–phenotype correlation of the X-linked Alport syndrome (XLAS) has been well elucidated in males, whereas it remains unclear in females. In this multicenter retrospective study, we analyzed the genotype–phenotype correlation in 216 Korean patients (male:female = 130:86) with XLAS between 2000 and 2021. The patients were divided into three groups according to their genotypes: the non-truncating group, the abnormal splicing group, and the truncating group. In male patients, approximately 60% developed kidney failure at the median age of 25.0 years, and kidney survival showed significant differences between the non-truncating and truncating groups (P < 0.001, hazard ratio (HR) 2.8) and splicing and truncating groups (P = 0.002, HR 3.1). Sensorineural hearing loss was detected in 65.1% of male patients, while hearing survival periods showed a highly significant difference between the non-truncating and truncating groups (P < 0.001, HR 5.1). In female patients, approximately 20% developed kidney failure at the median age of 50.2 years. The kidney survival was significantly different between the non-truncating and truncating groups (P = 0.006, HR 5.7). Our findings support the presence of genotype–phenotype correlation not only in male patients but also in female patients with XLAS. © 2023, The Author(s).
DOI
10.1038/s41598-023-34053-7
Appears in Collections:
의과대학 > 의학과 > Journal papers
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