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Alport syndrome is a hereditary disorder characterized by hematuria, proteinuria and progressive renal failure, frequently associated with extrarenal manifestations. The pathogenic variants of the COL4A5 gene are associated with X-linked Alport syndrome while those of the COL4A3 and COL4A4 genes are associated with the autosomal recessive (AR) or dominant (AD) form. The disease is characterized by considerable phenotypic variability linked to the different genes involved and the different mutations present, so the symptoms manifest themselves in different frequencies depending on the case. The existence of an autosomal dominant form of Alport syndrome has been identified in recent years thanks to next generation gene sequencing (NGS) techniques which have made it possible to highlight unknown genetic variants of Alport syndrome. The family studied by us presents concomitant heterozygous alterations of the COL4A3 genes (c.1029+5G>A with MAF 0 and c.3211-7A>G with MAF 1:100000), heterozygous alterations of the MTHFR gene (both C677T and A1298C) and homozygous alteration of the PAI-1 gene. While the variant c.3211-7A>G, as shown by genetic databases (ClinVar), appears to be benign, the intronic variant c.1029+5G>A (caused by exon skipping) can be classified as pathogenic due to its characteristics and the fact that it co-segregates with the phenotype within the family. The histological data, in one of the sisters, highlighted the presence of a discrete global glomerular sclerosis and the ultrastructural investigation a thinning of the glomerular basement membrane. New mutational variants of the COL4A3 gene may play a role as risk variants for the development of chronic kidney disease.
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http://dx.doi.org/10.69097/42-02-2025-07 | DOI Listing |
BMC Nephrol
September 2025
Cerrahpasa Medical Faculty, Division of Nephrology, Istanbul University-Cerrahpasa, Istanbul, Turkey.
Background: Immune complex-mediated membranoproliferative glomerulonephritis (IC-MPGN) and Alport syndrome are distinct glomerular diseases with different pathophysiologic mechanisms. Their coexistence is extremely rare and may present diagnostic and therapeutic challenges.
Case Presentation: A 42-year-old woman presented with persistent proteinuria and hematuria.
Intern Med
September 2025
Division of Nephrology and Hypertension, Department of Internal Medicine, The Jikei University School of Medicine, Japan.
Sci Rep
August 2025
Department of Nephrology & Rheumatology, Kunming Children's Hospital, Kunming Medical University, Kunming, Yunnan, 650228, China.
Alport syndrome is a hereditary glomerular disease driven by pathogenic variants in COL4A3-COL4A5 that compromise the α3-α4-α5 type IV collagen scaffold, manifesting as persistent hematuria, proteinuria, and ultimately end-stage renal disease. Its pronounced phenotypic variability, low sensitivity of renal biopsy, and limited response to ACE inhibitors complicate accurate diagnosis and therapy. In a cohort of 40 pedigrees from southwest China, we discovered 21 novel COL4A3-COL4A5 mutations.
View Article and Find Full Text PDFBiomedicines
August 2025
Department of Paediatrics and Adolescent Medicine, Faculty of Medicine, P. J. Šafárik University in Košice, 040 11 Košice, Slovakia.
Alport syndrome (AS) predominantly presents with X-linked inheritance worldwide. However, the epidemiological landscape remains poorly characterized, particularly among ethnic minority groups like the Roma minority in Slovakia. Our study aimed to investigate the inheritance patterns of AS in this region and determine whether a distinct pattern predominates.
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