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Erschienen in: Urolithiasis 1/2021

18.06.2020 | Original Paper

Characteristics of the genotype and phenotype in Chinese primary hyperoxaluria type 1 populations

verfasst von: Fangzhou Zhao, Jun Li, Lei Tang, Chunming Li, Wenying Wang, Chen Ning

Erschienen in: Urolithiasis | Ausgabe 1/2021

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Abstract

The aim of our study is to explore the relationship between genotype and phenotype in Chinese PH1 patients and determine the putative mutation hotspot regions. This was a retrospective study regarding 13 Chinese PH1 patients. And all sporadic published researches of Chinese PH1 populations were searched and enrolled based on the inclusive standard. All patients presented with multiple urolithiasis or nephrolithiasis. Urinary oxalate values demonstrated an obvious and extensive variability, ranging from 1.01 to 3.85 mmol/1.73 m2. Molecular diagnosis showed that 13 mutant types were detected. Infantile form patient (pt.) 10 and five patients (pts. 5, 7, 8, 9, 12) carrying c.815_816insGA or c.33_34insC demonstrated a worse prognosis, of whom pt. 5 progressed into ESRD 4 years later and died of chronic kidney failure. Based on the integrated Chinese mutation data, two variants (c.815_816insGA and c.33_34insC) were determined as the most common mutations. Besides, c.1049G>A was initially identified in a Chinese patient. Conclusions: heterogeneity between genotype and phenotype was observed and described in Chinese PH1 patients. c.815_816insGA and c.33_34insC which were recognized as AGXT mutation hotspot regions in China implied a poor prognosis. And c.1049G>A was not determined as the race-specific mutation of Pakistani.
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Metadaten
Titel
Characteristics of the genotype and phenotype in Chinese primary hyperoxaluria type 1 populations
verfasst von
Fangzhou Zhao
Jun Li
Lei Tang
Chunming Li
Wenying Wang
Chen Ning
Publikationsdatum
18.06.2020
Verlag
Springer Berlin Heidelberg
Erschienen in
Urolithiasis / Ausgabe 1/2021
Print ISSN: 2194-7228
Elektronische ISSN: 2194-7236
DOI
https://doi.org/10.1007/s00240-020-01201-x

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