Cargando…
Genetic Profile of Patients with Limb-Girdle Muscle Weakness in the Chilean Population
Hereditary myopathies are a group of genetically determined muscle disorders comprising more than 300 entities. In Chile, there are no specific registries of the distinct forms of these myopathies. We now report the genetic findings of a series of Chilean patients presenting with limb-girdle muscle...
Autores principales: | , , , , , , , , , , , , , , , , , , , , , , , |
---|---|
Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2022
|
Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9223019/ https://www.ncbi.nlm.nih.gov/pubmed/35741838 http://dx.doi.org/10.3390/genes13061076 |
_version_ | 1784733020429746176 |
---|---|
author | Cerino, Mathieu González-Hormazábal, Patricio Abaji, Mario Courrier, Sebastien Puppo, Francesca Mathieu, Yves Trangulao, Alejandra Earle, Nicholas Castiglioni, Claudia Díaz, Jorge Campero, Mario Hughes, Ricardo Vargas, Carmen Cortés, Rocío Kleinsteuber, Karin Acosta, Ignacio Urtizberea, J. Andoni Lévy, Nicolas Bartoli, Marc Krahn, Martin Jara, Lilian Caviedes, Pablo Gorokhova, Svetlana Bevilacqua, Jorge A. |
author_facet | Cerino, Mathieu González-Hormazábal, Patricio Abaji, Mario Courrier, Sebastien Puppo, Francesca Mathieu, Yves Trangulao, Alejandra Earle, Nicholas Castiglioni, Claudia Díaz, Jorge Campero, Mario Hughes, Ricardo Vargas, Carmen Cortés, Rocío Kleinsteuber, Karin Acosta, Ignacio Urtizberea, J. Andoni Lévy, Nicolas Bartoli, Marc Krahn, Martin Jara, Lilian Caviedes, Pablo Gorokhova, Svetlana Bevilacqua, Jorge A. |
author_sort | Cerino, Mathieu |
collection | PubMed |
description | Hereditary myopathies are a group of genetically determined muscle disorders comprising more than 300 entities. In Chile, there are no specific registries of the distinct forms of these myopathies. We now report the genetic findings of a series of Chilean patients presenting with limb-girdle muscle weakness of unknown etiology. Eighty-two patients were explored using high-throughput sequencing approaches with neuromuscular gene panels, establishing a definite genetic diagnosis in 49 patients (59.8%) and a highly probable genetic diagnosis in eight additional cases (9.8%). The most frequent causative genes identified were DYSF and CAPN3, accounting for 22% and 8.5% of the cases, respectively, followed by DMD (4.9%) and RYR1 (4.9%). The remaining 17 causative genes were present in one or two cases only. Twelve novel variants were identified. Five patients (6.1%) carried a variant of uncertain significance in genes partially matching the clinical phenotype. Twenty patients (24.4%) did not carry a pathogenic or likely pathogenic variant in the phenotypically related genes, including five patients (6.1%) presenting an autoimmune neuromuscular disorder. The relative frequency of the different forms of myopathy in Chile is like that of other series reported from different regions of the world with perhaps a relatively higher incidence of dysferlinopathy. |
format | Online Article Text |
id | pubmed-9223019 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
spelling | pubmed-92230192022-06-24 Genetic Profile of Patients with Limb-Girdle Muscle Weakness in the Chilean Population Cerino, Mathieu González-Hormazábal, Patricio Abaji, Mario Courrier, Sebastien Puppo, Francesca Mathieu, Yves Trangulao, Alejandra Earle, Nicholas Castiglioni, Claudia Díaz, Jorge Campero, Mario Hughes, Ricardo Vargas, Carmen Cortés, Rocío Kleinsteuber, Karin Acosta, Ignacio Urtizberea, J. Andoni Lévy, Nicolas Bartoli, Marc Krahn, Martin Jara, Lilian Caviedes, Pablo Gorokhova, Svetlana Bevilacqua, Jorge A. Genes (Basel) Article Hereditary myopathies are a group of genetically determined muscle disorders comprising more than 300 entities. In Chile, there are no specific registries of the distinct forms of these myopathies. We now report the genetic findings of a series of Chilean patients presenting with limb-girdle muscle weakness of unknown etiology. Eighty-two patients were explored using high-throughput sequencing approaches with neuromuscular gene panels, establishing a definite genetic diagnosis in 49 patients (59.8%) and a highly probable genetic diagnosis in eight additional cases (9.8%). The most frequent causative genes identified were DYSF and CAPN3, accounting for 22% and 8.5% of the cases, respectively, followed by DMD (4.9%) and RYR1 (4.9%). The remaining 17 causative genes were present in one or two cases only. Twelve novel variants were identified. Five patients (6.1%) carried a variant of uncertain significance in genes partially matching the clinical phenotype. Twenty patients (24.4%) did not carry a pathogenic or likely pathogenic variant in the phenotypically related genes, including five patients (6.1%) presenting an autoimmune neuromuscular disorder. The relative frequency of the different forms of myopathy in Chile is like that of other series reported from different regions of the world with perhaps a relatively higher incidence of dysferlinopathy. MDPI 2022-06-16 /pmc/articles/PMC9223019/ /pubmed/35741838 http://dx.doi.org/10.3390/genes13061076 Text en © 2022 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 Cerino, Mathieu González-Hormazábal, Patricio Abaji, Mario Courrier, Sebastien Puppo, Francesca Mathieu, Yves Trangulao, Alejandra Earle, Nicholas Castiglioni, Claudia Díaz, Jorge Campero, Mario Hughes, Ricardo Vargas, Carmen Cortés, Rocío Kleinsteuber, Karin Acosta, Ignacio Urtizberea, J. Andoni Lévy, Nicolas Bartoli, Marc Krahn, Martin Jara, Lilian Caviedes, Pablo Gorokhova, Svetlana Bevilacqua, Jorge A. Genetic Profile of Patients with Limb-Girdle Muscle Weakness in the Chilean Population |
title | Genetic Profile of Patients with Limb-Girdle Muscle Weakness in the Chilean Population |
title_full | Genetic Profile of Patients with Limb-Girdle Muscle Weakness in the Chilean Population |
title_fullStr | Genetic Profile of Patients with Limb-Girdle Muscle Weakness in the Chilean Population |
title_full_unstemmed | Genetic Profile of Patients with Limb-Girdle Muscle Weakness in the Chilean Population |
title_short | Genetic Profile of Patients with Limb-Girdle Muscle Weakness in the Chilean Population |
title_sort | genetic profile of patients with limb-girdle muscle weakness in the chilean population |
topic | Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9223019/ https://www.ncbi.nlm.nih.gov/pubmed/35741838 http://dx.doi.org/10.3390/genes13061076 |
work_keys_str_mv | AT cerinomathieu geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT gonzalezhormazabalpatricio geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT abajimario geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT courriersebastien geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT puppofrancesca geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT mathieuyves geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT trangulaoalejandra geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT earlenicholas geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT castiglioniclaudia geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT diazjorge geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT camperomario geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT hughesricardo geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT vargascarmen geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT cortesrocio geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT kleinsteuberkarin geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT acostaignacio geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT urtizbereajandoni geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT levynicolas geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT bartolimarc geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT krahnmartin geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT jaralilian geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT caviedespablo geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT gorokhovasvetlana geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation AT bevilacquajorgea geneticprofileofpatientswithlimbgirdlemuscleweaknessinthechileanpopulation |