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dc.contributor.author
Buonfiglio, Paula Inés  
dc.contributor.author
Menazzi, Sebastián  
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Francipane, Liliana  
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Lotersztein, Vanesa  
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Ferreiro, Verónica  
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Elgoyhen, Ana Belen  
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Dalamon, Viviana Karina  
dc.date.available
2024-03-07T14:48:49Z  
dc.date.issued
2023-02  
dc.identifier.citation
Buonfiglio, Paula Inés; Menazzi, Sebastián; Francipane, Liliana; Lotersztein, Vanesa; Ferreiro, Verónica; et al.; Mitochondrial DNA variants in a cohort from Argentina with suspected Leber’s hereditary optic neuropathy (LHON); Public Library of Science; Plos One; 18; 2; 2-2023; 1-17  
dc.identifier.issn
1932-6203  
dc.identifier.uri
http://hdl.handle.net/11336/229734  
dc.description.abstract
The present study investigates the spectrum and analysis of mitochondrial DNA (mtDNA) variants associated with Leber hereditary optic neuropathy (LHON) in an Argentinean cohort, analyzing 3 LHON-associated mitochondrial genes. In 32% of the cases, molecular confirmation of the diagnosis could be established, due to the identification of disease-causing variants. A total of 54 variants were observed in a cohort of 100 patients tested with direct sequencing analysis. The frequent causative mutations m.11778G>A in MT-ND4, m.3460G>A in MT-ND1, and m.14484T>C in MT-ND6 were identified in 28% of the cases of our cohort. Secondary mutations in this Argentinean LHON cohort were m.11253T>C p.Ile165Thr in MT-ND4, identified in three patients (3/100, 3%) and m.3395A>G p.Tyr30Cys in MT-ND1, in one of the patients studied (1%). This study shows, for the first time, the analysis of mtDNA variants in patients with a probable diagnosis of LHON in Argentina. Standard molecular methods are an effective first approach in order to achieve genetic diagnosis of the disease, leaving NGS tests for those patients with negative results.  
dc.format
application/pdf  
dc.language.iso
eng  
dc.publisher
Public Library of Science  
dc.rights
info:eu-repo/semantics/openAccess  
dc.rights.uri
https://creativecommons.org/licenses/by-nc-sa/2.5/ar/  
dc.subject
LEBER  
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LHON  
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MITOCHONDRIAL VARIANTS  
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GENETIC DIAGNOSIS  
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Genética Humana  
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Medicina Básica  
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CIENCIAS MÉDICAS Y DE LA SALUD  
dc.title
Mitochondrial DNA variants in a cohort from Argentina with suspected Leber’s hereditary optic neuropathy (LHON)  
dc.type
info:eu-repo/semantics/article  
dc.type
info:ar-repo/semantics/artículo  
dc.type
info:eu-repo/semantics/publishedVersion  
dc.date.updated
2024-02-22T13:01:51Z  
dc.journal.volume
18  
dc.journal.number
2  
dc.journal.pagination
1-17  
dc.journal.pais
Estados Unidos  
dc.description.fil
Fil: Buonfiglio, Paula Inés. Consejo Nacional de Investigaciones Científicas y Técnicas. Instituto de Investigaciones en Ingeniería Genética y Biología Molecular "Dr. Héctor N. Torres"; Argentina  
dc.description.fil
Fil: Menazzi, Sebastián. Universidad de Buenos Aires. Facultad de Medicina. Hospital de Clínicas General San Martín; Argentina  
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Fil: Francipane, Liliana. Universidad de Buenos Aires. Facultad de Medicina. Hospital de Clínicas General San Martín; Argentina  
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Fil: Lotersztein, Vanesa. Gobierno de la Ciudad Autónoma de Buenos Aires. Hospital General de Agudos Doctor Cosme Argerich; Argentina  
dc.description.fil
Fil: Ferreiro, Verónica. No especifíca;  
dc.description.fil
Fil: Elgoyhen, Ana Belen. Consejo Nacional de Investigaciones Científicas y Técnicas. Instituto de Investigaciones en Ingeniería Genética y Biología Molecular "Dr. Héctor N. Torres"; Argentina. Universidad de Buenos Aires. Facultad de Medicina. Hospital de Clínicas General San Martín; Argentina. Gobierno de la Ciudad Autónoma de Buenos Aires. Hospital General de Agudos Doctor Cosme Argerich; Argentina  
dc.description.fil
Fil: Dalamon, Viviana Karina. Consejo Nacional de Investigaciones Científicas y Técnicas. Instituto de Investigaciones en Ingeniería Genética y Biología Molecular "Dr. Héctor N. Torres"; Argentina  
dc.journal.title
Plos One  
dc.relation.alternativeid
info:eu-repo/semantics/altIdentifier/doi/http://dx.doi.org/10.1371/journal.pone.0275703