Evidence map›Paper›PMID 41137992›Full record

ArticleJournal of inherited metabolic disease2025

Transforming Growth Factor-β-Mediated Fibrotic Remodeling Drives Chronic Kidney Disease in Methylmalonic Aciduria and Propionic Aciduria-Identification of a New Therapeutic Target.

Karina A Zeyer, Stefan Tholen, Oliver Schilling, Leonie Gerling, Marina Morath, Stefan Kölker, Alexander Nyström, Ute Spiekerkoetter, Anke Schumann

Abstract read
In one paragraph

Article in Journal of inherited metabolic disease, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

9 authors.

Karina A ZeyerDepartment of Pediatrics, Adolescent Medicine and Neonatology, Faculty of Medicine, Medical Center - University of Freiburg, Freiburg, Germany.ORCID https://orcid.org/0009-0007-0640-2528
Stefan TholenInstitute for Surgical Pathology, Medical Center - University of Freiburg/Medical Faculty - University of Freiburg, Freiburg, Germany.
Oliver SchillingInstitute for Surgical Pathology, Medical Center - University of Freiburg/Medical Faculty - University of Freiburg, Freiburg, Germany.
Leonie GerlingDepartment of Pediatrics, Adolescent Medicine and Neonatology, Faculty of Medicine, Medical Center - University of Freiburg, Freiburg, Germany.
Marina MorathCenter for Pediatric and Adolescent Medicine, Division of Pediatric Neurology and Metabolic Medicine, Heidelberg University, Medical Faculty of Heidelberg, Heidelberg University Hospital, Heidelberg, Germany.
Stefan KölkerCenter for Pediatric and Adolescent Medicine, Division of Pediatric Neurology and Metabolic Medicine, Heidelberg University, Medical Faculty of Heidelberg, Heidelberg University Hospital, Heidelberg, Germany.
Alexander NyströmDepartment of Dermatology, Faculty of Medicine, Medical Center - University of Freiburg, Freiburg, Germany.ORCID https://orcid.org/0000-0002-4666-2240
Ute SpiekerkoetterDepartment of Pediatrics, Adolescent Medicine and Neonatology, Faculty of Medicine, Medical Center - University of Freiburg, Freiburg, Germany.
Anke SchumannDepartment of Pediatrics, Adolescent Medicine and Neonatology, Faculty of Medicine, Medical Center - University of Freiburg, Freiburg, Germany.

Funding

Berta-Ottenstein-Programme for Advanced Clinician Scientists, Faculty of Medicine, University of FreiburgBerta-Ottenstein-Programme for Clinician Scientists, Faculty of Medicine, University of FreiburgDeutsche Forschungsgemeinschaft (DFG, German Research Foundation) 431984000 - CRC 1453 NephrogeneticsDivision of Inborn Errors of Metabolism at the Department of Pediatrics, Adolescent Medicine, and Neonatology FreiburgFreiburg Center for Rare DiseasesMedical Faculty of the University of Freiburg 2021/A3-SchMedical Faculty of the University of Freiburg 2023/A3-Sch
6 · The paper itself

Abstract

Propionic aciduria (PA-uria) and methylmalonic aciduria (MMA-uria) are caused by defects in propionate catabolism. While chronic kidney disease (CKD) is a well-established complication in MMA-uria, renal involvement in PA-uria has only come into focus more recently, and the underlying mechanisms remain poorly understood. We investigated human renal epithelial cells from patients with PA-uria, MMA-uria, and healthy controls under metabolic stress, induced by methylmalonic acid, methylcitric acid, high-protein, or isoleucine/valine-enriched media. Proteomic profiling revealed significant enrichment of extracellular matrix (ECM)-related pathways in PA-uria cells. Both PA-uria and MMA-uria cells exhibited increased deposition of fibronectin and collagen fibers, which were further amplified under metabolic stress conditions. Transforming growth factor beta (TGF-β) signaling was identified as a key pro-fibrotic pathway. Pharmacological inhibition of the TGF-β receptor signaling normalized fibronectin and collagen deposition in both PA-uria and MMA-uria cells. Treatment with losartan, an angiotensin II type 1 receptor blocker known to modulate TGF-β signaling, also reversed the enhanced ECM deposition. This is the first study to mechanistically link ECM remodeling and TGF-β signaling to CKD pathogenesis in both PA-uria and MMA-uria. Our findings highlight fibrotic remodeling as a shared pathogenic feature and suggest that losartan, a widely available and well-tolerated drug, could be repurposed to mitigate renal fibrosis in these disorders.

Indexed as

Amino Acid Metabolism, Inborn ErrorsPropionic AcidemiaRenal Insufficiency, ChronicTransforming Growth Factor betaCollagenEpithelial CellsExtracellular MatrixFemaleFibronectinsFibrosisHumansKidneyLosartanMalePropionatesProteomicsCollagenFibronectinsLosartanPropionatespropionic acidTransforming Growth Factor beta

Identifiers

PMID41137992
PMCPMC12553402

What OpenQuestion holds

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LicenceCC BY
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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.