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DOI 10.1038/s41598-020-79331-w

Медицинская библиотека · Scientific reports · 2021

Autosomal dominant tubulointerstitial kidney disease genotype and phenotype correlation in a Chinese cohort.

Gong Kunjing, Xia Min, Wang Yaqin, Wang Na, Liu Ying, Zhang Victor Wei, Cheng Hong, Chen Yuqing

СтатьяПоддержка исследования: вне правительства СШАПеревод в очереди

Аннотация

Genes of UMOD, HNF1B, MUC1, REN and SEC61A1 were reported to be associated with autosomal dominant tubulointerstitial kidney disease (ADTKD). 48 probands and their family members (N = 27) were enrolled in this genetic screening study. A combination of methods was employed for comprehensive molecular analysis of both copy number variations (CNVs) and single nucleotide variants (SNVs). 35 probands were followed for years. The phenotype-genotype and genotype-outcome correlation were inferred from these datasets. In this cohort, 18 probands were diagnosed with ADTKD, according to Kidney Disease: Improving Global Outcomes (KDIGO) guideline. Moreover, 11 probands were diagnosed with ADTKD-UMOD, one with ADTKD-REN and one with ADTKD-HNF1B, based on molecularly confirmed pathogenic variants. The 11 UMOD variants were mainly located in codons 28 to 289 and half of the variants were found to change the cysteine amino acid. According to the follow-up data, suspected ADTKD individuals had a better prognosis compared to ADTKD individuals (p = 0.029). Individuals with a cysteine substitution in the UMOD gene appeared to have a better prognosis than individuals with other amino acid substitutions (p = 0.015).

Для цитирования: Gong Kunjing, Xia Min, Wang Yaqin, Wang Na, Liu Ying, Zhang Victor Wei, Cheng Hong, Chen Yuqing. Autosomal dominant tubulointerstitial kidney disease genotype and phenotype correlation in a Chinese cohort.. Scientific reports. 2021. DOI: 10.1038/s41598-020-79331-w.

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