Cargando…

High Rate of Mutations of Adhesion Molecules and Extracellular Matrix Glycoproteins in Patients with Adult-Onset Focal and Segmental Glomerulosclerosis

(1) Background: Focal and segmental glomerulosclerosis (FSGS) is a pattern of injury that results from podocyte loss in the setting of a wide variety of injurious mechanisms. These include both acquired and genetic as well as primary and secondary causes, or a combination thereof, without optimal th...

Descripción completa

Detalles Bibliográficos
Autores principales: Marcos González, Sara, Rodrigo Calabia, Emilio, Varela, Ignacio, Červienka, Michal, Freire Salinas, Javier, Gómez Román, José Javier
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10296603/
https://www.ncbi.nlm.nih.gov/pubmed/37371859
http://dx.doi.org/10.3390/biomedicines11061764
_version_ 1785063689095741440
author Marcos González, Sara
Rodrigo Calabia, Emilio
Varela, Ignacio
Červienka, Michal
Freire Salinas, Javier
Gómez Román, José Javier
author_facet Marcos González, Sara
Rodrigo Calabia, Emilio
Varela, Ignacio
Červienka, Michal
Freire Salinas, Javier
Gómez Román, José Javier
author_sort Marcos González, Sara
collection PubMed
description (1) Background: Focal and segmental glomerulosclerosis (FSGS) is a pattern of injury that results from podocyte loss in the setting of a wide variety of injurious mechanisms. These include both acquired and genetic as well as primary and secondary causes, or a combination thereof, without optimal therapy, and a high rate of patients develop end-stage renal disease (ESRD). Genetic studies have helped improve the global understanding of FSGS syndrome; thus, we hypothesize that patients with primary FSGS may have underlying alterations in adhesion molecules or extracellular matrix glycoproteins related to previously unreported mutations that may be studied through next-generation sequencing (NGS). (2) Methods: We developed an NGS panel with 29 genes related to adhesion and extracellular matrix glycoproteins. DNA was extracted from twenty-three FSGS patients diagnosed by renal biopsy; (3) Results: The average number of accumulated variants in FSGS patients was high. We describe the missense variant ITGB3c.1199G>A, which is considered pathogenic; in addition, we discovered the nonsense variant CDH1c.499G>T, which lacks a Reference SNP (rs) Report and is considered likely pathogenic. (4) Conclusions: To the best of our knowledge, this is the first account of a high rate of change in extracellular matrix glycoproteins and adhesion molecules in individuals with adult-onset FSGS. The combined effect of all these variations may result in a genotype that is vulnerable to the pathogenesis of glomerulopathy.
format Online
Article
Text
id pubmed-10296603
institution National Center for Biotechnology Information
language English
publishDate 2023
publisher MDPI
record_format MEDLINE/PubMed
spelling pubmed-102966032023-06-28 High Rate of Mutations of Adhesion Molecules and Extracellular Matrix Glycoproteins in Patients with Adult-Onset Focal and Segmental Glomerulosclerosis Marcos González, Sara Rodrigo Calabia, Emilio Varela, Ignacio Červienka, Michal Freire Salinas, Javier Gómez Román, José Javier Biomedicines Article (1) Background: Focal and segmental glomerulosclerosis (FSGS) is a pattern of injury that results from podocyte loss in the setting of a wide variety of injurious mechanisms. These include both acquired and genetic as well as primary and secondary causes, or a combination thereof, without optimal therapy, and a high rate of patients develop end-stage renal disease (ESRD). Genetic studies have helped improve the global understanding of FSGS syndrome; thus, we hypothesize that patients with primary FSGS may have underlying alterations in adhesion molecules or extracellular matrix glycoproteins related to previously unreported mutations that may be studied through next-generation sequencing (NGS). (2) Methods: We developed an NGS panel with 29 genes related to adhesion and extracellular matrix glycoproteins. DNA was extracted from twenty-three FSGS patients diagnosed by renal biopsy; (3) Results: The average number of accumulated variants in FSGS patients was high. We describe the missense variant ITGB3c.1199G>A, which is considered pathogenic; in addition, we discovered the nonsense variant CDH1c.499G>T, which lacks a Reference SNP (rs) Report and is considered likely pathogenic. (4) Conclusions: To the best of our knowledge, this is the first account of a high rate of change in extracellular matrix glycoproteins and adhesion molecules in individuals with adult-onset FSGS. The combined effect of all these variations may result in a genotype that is vulnerable to the pathogenesis of glomerulopathy. MDPI 2023-06-20 /pmc/articles/PMC10296603/ /pubmed/37371859 http://dx.doi.org/10.3390/biomedicines11061764 Text en © 2023 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
spellingShingle Article
Marcos González, Sara
Rodrigo Calabia, Emilio
Varela, Ignacio
Červienka, Michal
Freire Salinas, Javier
Gómez Román, José Javier
High Rate of Mutations of Adhesion Molecules and Extracellular Matrix Glycoproteins in Patients with Adult-Onset Focal and Segmental Glomerulosclerosis
title High Rate of Mutations of Adhesion Molecules and Extracellular Matrix Glycoproteins in Patients with Adult-Onset Focal and Segmental Glomerulosclerosis
title_full High Rate of Mutations of Adhesion Molecules and Extracellular Matrix Glycoproteins in Patients with Adult-Onset Focal and Segmental Glomerulosclerosis
title_fullStr High Rate of Mutations of Adhesion Molecules and Extracellular Matrix Glycoproteins in Patients with Adult-Onset Focal and Segmental Glomerulosclerosis
title_full_unstemmed High Rate of Mutations of Adhesion Molecules and Extracellular Matrix Glycoproteins in Patients with Adult-Onset Focal and Segmental Glomerulosclerosis
title_short High Rate of Mutations of Adhesion Molecules and Extracellular Matrix Glycoproteins in Patients with Adult-Onset Focal and Segmental Glomerulosclerosis
title_sort high rate of mutations of adhesion molecules and extracellular matrix glycoproteins in patients with adult-onset focal and segmental glomerulosclerosis
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10296603/
https://www.ncbi.nlm.nih.gov/pubmed/37371859
http://dx.doi.org/10.3390/biomedicines11061764
work_keys_str_mv AT marcosgonzalezsara highrateofmutationsofadhesionmoleculesandextracellularmatrixglycoproteinsinpatientswithadultonsetfocalandsegmentalglomerulosclerosis
AT rodrigocalabiaemilio highrateofmutationsofadhesionmoleculesandextracellularmatrixglycoproteinsinpatientswithadultonsetfocalandsegmentalglomerulosclerosis
AT varelaignacio highrateofmutationsofadhesionmoleculesandextracellularmatrixglycoproteinsinpatientswithadultonsetfocalandsegmentalglomerulosclerosis
AT cervienkamichal highrateofmutationsofadhesionmoleculesandextracellularmatrixglycoproteinsinpatientswithadultonsetfocalandsegmentalglomerulosclerosis
AT freiresalinasjavier highrateofmutationsofadhesionmoleculesandextracellularmatrixglycoproteinsinpatientswithadultonsetfocalandsegmentalglomerulosclerosis
AT gomezromanjosejavier highrateofmutationsofadhesionmoleculesandextracellularmatrixglycoproteinsinpatientswithadultonsetfocalandsegmentalglomerulosclerosis