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0000000000 65535 f 0000000000 65535 f trailer <<6109564AFD1B944593AEE7BD6F86FE3D>]>> startxref 116 %%EOF Estudio genético de hipercolesterolemia familiar en una población hospitalaria de la Ciudad de Buenos Aires - Archivo PPCT

Estudio genético de hipercolesterolemia familiar en una población hospitalaria de la Ciudad de Buenos Aires;
Genetic Testing of Familial Hypercholesterolemia in a Hospital-Based Population of the City of Buenos Aires

Creators:Gómez, Andrea, Helman, Lorena, Giunta, Gustavo, Toscanini, Ulises, Cuniberti, Luis
2018-06-26

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Descripción

Introducción: La hipercolesterolemia familiar es una hiperlipidemia primaria. Se trata de un trastorno genético autosómico dominante del metabolismo de las lipoproteínas, caracterizado por concentraciones plasmáticas elevadas de colesterol unido a lipoproteínas de baja densidad y presencia de xantomas tendinosos, y está asociado con el desarrollo prematuro de enfermedad cardiovascular. Objetivos: Investigar la presencia de mutaciones en el principal gen asociado al desarrollo de hipercolesterolemia familiar (LDLR) en un grupo de pacientes identificados como “casos índices”, de entre aquellos que concurren al Servicio de Lípidos del Hospital Universitario Fundación Favaloro con diagnóstico clínico de hipercolesterolemia familiar. Determinar la composición ancestral de la población estudiada. Material y métodos: Se estudió una población de 38 pacientes con diagnóstico clínico de hipercolesterolemia familiar. La región codificante y las zonas intrónicas adyacentes del gen LDLR se secuenciaron automáticamente por el método de Sanger. Se investigó el componente ancestral de la población estudiada a partir del análisis de 46 marcadores informativos de ancestralidad (AIM-Indel). Resultados: Se identificaron 50 variantes diferentes, de las cuales el 48% se consideraron patogénicas. Se logró establecer una correlación genotipo-gravedad del fenotipo en el 60,5% de los pacientes estudiados. El componente ancestral de la población estudiada fue predominantemente europeo, seguido de un componente nativo-americano y, en menor proporción, africano. Conclusiones: El análisis genético por secuenciación del gen LDLR en pacientes identificados como “casos índices” con diagnóstico clínico de hipercolesterolemia familiar permite correlacionar el dato genético con la gravedad del fenotipo observado clínicamente y efectuar un diagnóstico en cascada en los miembros de la familia que presentan los criterios de inclusión considerados.

Background: Familial hypercholesterolemia is a primary hyperlipidemia. It is an autosomal dominant genetic disorder of lipoprotein metabolism, characterized by elevated plasma low-density lipoprotein cholesterol and presence of tendon xanthomas, and is associated with early cardiovascular disease. Objectives: The aim of this study was to investigate the presence of mutations in the main gene associated with the development of familial hypercholesterolemia (LDLR) in a group of patients identified as “index cases” attending the Lipid Clinic of the Hospital Universitario Fundación Favaloro with clinical diagnosis of familial hypercholesterolemia. The ancestral composition of the study population was determined. Methods: We evaluated 38 patients with clinical diagnosis of familial hypercholesterolemia. Mutation screening of the LDLR gene coding regions and adjacent intronic areas was performed using Sanger sequencing. The ancestral component of the study population was investigated using 46 ancestry inference markers (AIM-Indel). Results: Fifty different variants were identified, 48% of which were considered pathogenic. A genotype-phenotype severity correlation was established in 60.5% of the patients evaluated. The ancestral component of the study population was predominantly European, followed by native-American and African in lower proportion. Conclusions: Genetic testing by LDLR gene sequencing in patients identified as “index cases” with clinical diagnosis of familial hypercholesterolemia allows the correlation between the genetic information and the severity of the clinical phenotype to a cascade testing of the family members presenting the inclusion criteria considered.

Metadatos destacados

Colecciones
Argentine Journal of Cardiology

Editor

Sociedad Argentina de Cardiología

Fuente

Revista Argentina de Cardiología; Vol 86, No 2 (2018); 103-109, Argentine Journal of Cardiology; Vol 86, No 2 (2018); 103-109

Citación

Gómez, Andrea et al., “Estudio genético de hipercolesterolemia familiar en una población hospitalaria de la Ciudad de Buenos Aires,” Archivo PPCT, consulta 4 de abril de 2026, http://archivoppct.caicyt.gov.ar/items/show/9373.

Dublin Core

Autor

Gómez, Andrea
Helman, Lorena
Giunta, Gustavo
Toscanini, Ulises
Cuniberti, Luis

Fuente

Revista Argentina de Cardiología; Vol 86, No 2 (2018); 103-109
Argentine Journal of Cardiology; Vol 86, No 2 (2018); 103-109

Editor

Sociedad Argentina de Cardiología

Fecha

2018-06-26

Derechos

Los que firman al pié, certificamos que tenemos total responsabilidad por la conducción de este estudio y por el diseño y la interpretación de los datos. Nosotros escribimos el manuscrito y somos responsables por la decisión acerca del mismo. Cada uno de nosotros cumple la definición de autor como se afirma en el Comité Internacional de Editores de Revistas Médicas (International Committee of Medical Journal Editors, ver www.icmje.org). Nosotros hemos visto y aprobado el manuscrito final. Ni el artículo, ni ninguna parte esencial del mismo, incluido las tablas y las figuras, será publicado o admitido para arbitrar a otra parte antes de aparecer en la Revista.También notificamos haber leído la sección “conflicto de intereses”, y revelaríamos cualquiera que existiera. Dejamos constancia que si nuestro artículo se publicara en la RAC, cederíamos los derechos (copyright) a la Revista.Los documentos publicados en esta revista están bajo la licencia Creative Commons Atribución-NoComercial-Compartir-Igual 2.5 Argentina.
Those signing below certify that we have full responsibility for the conduction of this study and for the design and interpretation of the information. We wrote the manuscript and are responsible for its decision. Each of us fulfills the definition of authorship as stated by the International Committee of Medical Journal Editors ( www.icmje.org). We have signed and approved the final manuscript. Neither the manuscript, nor any essential part thereof, including tables and figures, will be published or accepted for refereeing elsewhere before being published in the Journal. We have also read the "Conflict of Interest" section and would disclose any existing. We state that if our manuscript is published in the RAC, we shall transfer the copyright to the Journal.

Formato

Idioma

spa
eng

Tipo

info:eu-repo/semantics/article
info:eu-repo/semantics/publishedVersion