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...

Descripción completa

Detalles Bibliográficos
Autores principales: 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.
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