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.
Transforming Growth Factor-β-Mediated Fibrotic Remodeling Drives Chronic Kidney Disease in Methylmalonic Aciduria and Propionic Aciduria-Identification of a New Therapeutic Target.
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作者:Zeyer Karina A, Tholen Stefan, Schilling Oliver, Gerling Leonie, Morath Marina, Kölker Stefan, Nyström Alexander, Spiekerkoetter Ute, Schumann Anke
| 期刊: | Journal of Inherited Metabolic Disease | 影响因子: | 3.800 |
| 时间: | 2025 | 起止号: | 2025 Nov;48(6):e70111 |
| doi: | 10.1002/jimd.70111 | ||
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