A non-coding signature in SHROOM3 is associated with kidney disease progression in Fabry disease

Fabry disease is a rare, X-linked lysosomal storage disorder that often leads to progressive kidney dysfunction. Despite carrying the same pathogenic GLA variant, patients exhibit considerable variability in the onset and progression of Fabry nephropathy, suggesting the involvement of additional genetic modifiers. This study aimed to investigate the possible role of genetic polymorphisms in non-coding regions. A total of 284 patients with Fabry disease were included in the study and divided into two groups based on the progression of the kidney disease. Ten selected single nucleotide polymorphisms located in non-coding regions of podocyte-related genes were analyzed using quantitative PCR with TaqMan probes. The analysis revealed significant associations between specific genotypes and an increased risk of rapid progression of Fabry nephropathy. In particular, the rs9992101 and rs17319721 polymorphisms in the SHROOM3 gene were significantly associated with higher odds of accelerated kidney function decline. However, neither of these polymorphisms nor the polygenic risk scores were associated with conventional biomarkers of kidney disease. Our results suggest that non-coding genetic variants in podocyte-related genes may contribute to the phenotypic variability observed in Fabry nephropathy. The integration of such genetic biomarkers into clinical practice could improve early risk stratification, support more individualized patient monitoring, and facilitate therapeutic decision-making.

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Publication Details

Journal
Molecular Genetics and Metabolism
Published
2025-12-25
DOI
https://doi.org/10.1016/j.ymgme.2025.109710
Primary Topic
Lysosomal Storage Disorders Research
Type
article
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article

A non-coding signature in SHROOM3 is associated with kidney disease progression in Fabry disease

Tina Levstek, Gheona Altarescu, Gabriela Dostálová, Albina Nowak et al.
Molecular Genetics and Metabolism
Lysosomal Storage Disorders Research
article

A non-coding signature in SHROOM3 is associated with kidney disease progression in Fabry disease

Tina Levstek, Gheona Altarescu, Gabriela Dostálová, Albina Nowak, Bojan Vujkovac, Markéta Šafaříková, Nika Breznik, Kaja Balant Marin, Aleš Linhart, Tisa Podkrajšek, Katarina Trebušak Podkrajšek, João-Paulo Oliveira
article en

Abstract

Fabry disease is a rare, X-linked lysosomal storage disorder that often leads to progressive kidney dysfunction. Despite carrying the same pathogenic GLA variant, patients exhibit considerable variability in the onset and progression of Fabry nephropathy, suggesting the involvement of additional genetic modifiers. This study aimed to investigate the possible role of genetic polymorphisms in non-coding regions. A total of 284 patients with Fabry disease were included in the study and divided into two groups based on the progression of the kidney disease. Ten selected single nucleotide polymorphisms located in non-coding regions of podocyte-related genes were analyzed using quantitative PCR with TaqMan probes. The analysis revealed significant associations between specific genotypes and an increased risk of rapid progression of Fabry nephropathy. In particular, the rs9992101 and rs17319721 polymorphisms in the SHROOM3 gene were significantly associated with higher odds of accelerated kidney function decline. However, neither of these polymorphisms nor the polygenic risk scores were associated with conventional biomarkers of kidney disease. Our results suggest that non-coding genetic variants in podocyte-related genes may contribute to the phenotypic variability observed in Fabry nephropathy. The integration of such genetic biomarkers into clinical practice could improve early risk stratification, support more individualized patient monitoring, and facilitate therapeutic decision-making.

Molecular Genetics and MetabolismVol. 147(1)
University of Ljubljana (SI), Charles University (CZ), Ljubljana University Medical Centre (SI), Shaare Zedek Medical Center (IL), Hospital de São João (PT), University Hospital of Zurich (CH), CUF Porto Hospital (PT), Šolski center Slovenj Gradec (SI), General University Hospital in Prague (CZ)
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Openalex Percentile: Top 33%
Lysosomal Storage Disorders Research
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