Cargando…

Clinical predictors of a positive genetic test in hypertrophic cardiomyopathy in the Brazilian population

BACKGROUND: Hypertrophic cardiomyopathy is a genetic autosomal dominant disease characterized by left ventricular hypertrophy. The molecular diagnosis is important but still expensive. This work aimed to find clinical predictors of a positive genetic test in a Brazilian tertiary centre cohort of ind...

Descripción completa

Detalles Bibliográficos
Autores principales: Marsiglia, Julia Daher Carneiro, Credidio, Flávia Laghi, de Oliveira, Théo Gremen Mimary, Reis, Rafael Ferreira, Antunes, Murillo de Oliveira, de Araujo, Aloir Queiroz, Pedrosa, Rodrigo Pinto, Barbosa-Ferreira, João Marcos Bemfica, Mady, Charles, Krieger, José Eduardo, Arteaga-Fernandez, Edmundo, Pereira, Alexandre Costa
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2014
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3995628/
https://www.ncbi.nlm.nih.gov/pubmed/24625281
http://dx.doi.org/10.1186/1471-2261-14-36
_version_ 1782312902877773824
author Marsiglia, Julia Daher Carneiro
Credidio, Flávia Laghi
de Oliveira, Théo Gremen Mimary
Reis, Rafael Ferreira
Antunes, Murillo de Oliveira
de Araujo, Aloir Queiroz
Pedrosa, Rodrigo Pinto
Barbosa-Ferreira, João Marcos Bemfica
Mady, Charles
Krieger, José Eduardo
Arteaga-Fernandez, Edmundo
Pereira, Alexandre Costa
author_facet Marsiglia, Julia Daher Carneiro
Credidio, Flávia Laghi
de Oliveira, Théo Gremen Mimary
Reis, Rafael Ferreira
Antunes, Murillo de Oliveira
de Araujo, Aloir Queiroz
Pedrosa, Rodrigo Pinto
Barbosa-Ferreira, João Marcos Bemfica
Mady, Charles
Krieger, José Eduardo
Arteaga-Fernandez, Edmundo
Pereira, Alexandre Costa
author_sort Marsiglia, Julia Daher Carneiro
collection PubMed
description BACKGROUND: Hypertrophic cardiomyopathy is a genetic autosomal dominant disease characterized by left ventricular hypertrophy. The molecular diagnosis is important but still expensive. This work aimed to find clinical predictors of a positive genetic test in a Brazilian tertiary centre cohort of index cases with HCM. METHODS: In the study were included patients with HCM clinical diagnosis. For genotype x phenotype comparison we have evaluated echocardiographic, electrocardiographic, and nuclear magnetic resonance measures. All patients answered a questionnaire about familial history of HCM and/or sudden death. β-myosin heavy chain, myosin binding protein C, and troponin T genes were sequenced for genetic diagnosis. RESULTS: The variables related to a higher probability of a positive genetic test were familial history of HCM, higher mean heart frequency, presence of NSVT and lower age. Probabilities of having a positive molecular genetic test were calculated from the final multivariate logistic regression model and were used to identify those with a higher probability of a positive molecular diagnosis. CONCLUSIONS: We developed an easy and fast screening method that takes into account only clinical data that can help to select the patients with a high probability of positive genetic results from molecular sequencing of Brazilian HCM patients.
format Online
Article
Text
id pubmed-3995628
institution National Center for Biotechnology Information
language English
publishDate 2014
publisher BioMed Central
record_format MEDLINE/PubMed
spelling pubmed-39956282014-05-07 Clinical predictors of a positive genetic test in hypertrophic cardiomyopathy in the Brazilian population Marsiglia, Julia Daher Carneiro Credidio, Flávia Laghi de Oliveira, Théo Gremen Mimary Reis, Rafael Ferreira Antunes, Murillo de Oliveira de Araujo, Aloir Queiroz Pedrosa, Rodrigo Pinto Barbosa-Ferreira, João Marcos Bemfica Mady, Charles Krieger, José Eduardo Arteaga-Fernandez, Edmundo Pereira, Alexandre Costa BMC Cardiovasc Disord Research Article BACKGROUND: Hypertrophic cardiomyopathy is a genetic autosomal dominant disease characterized by left ventricular hypertrophy. The molecular diagnosis is important but still expensive. This work aimed to find clinical predictors of a positive genetic test in a Brazilian tertiary centre cohort of index cases with HCM. METHODS: In the study were included patients with HCM clinical diagnosis. For genotype x phenotype comparison we have evaluated echocardiographic, electrocardiographic, and nuclear magnetic resonance measures. All patients answered a questionnaire about familial history of HCM and/or sudden death. β-myosin heavy chain, myosin binding protein C, and troponin T genes were sequenced for genetic diagnosis. RESULTS: The variables related to a higher probability of a positive genetic test were familial history of HCM, higher mean heart frequency, presence of NSVT and lower age. Probabilities of having a positive molecular genetic test were calculated from the final multivariate logistic regression model and were used to identify those with a higher probability of a positive molecular diagnosis. CONCLUSIONS: We developed an easy and fast screening method that takes into account only clinical data that can help to select the patients with a high probability of positive genetic results from molecular sequencing of Brazilian HCM patients. BioMed Central 2014-03-13 /pmc/articles/PMC3995628/ /pubmed/24625281 http://dx.doi.org/10.1186/1471-2261-14-36 Text en Copyright © 2014 Marsiglia et al.; licensee BioMed Central Ltd. http://creativecommons.org/licenses/by/2.0 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Research Article
Marsiglia, Julia Daher Carneiro
Credidio, Flávia Laghi
de Oliveira, Théo Gremen Mimary
Reis, Rafael Ferreira
Antunes, Murillo de Oliveira
de Araujo, Aloir Queiroz
Pedrosa, Rodrigo Pinto
Barbosa-Ferreira, João Marcos Bemfica
Mady, Charles
Krieger, José Eduardo
Arteaga-Fernandez, Edmundo
Pereira, Alexandre Costa
Clinical predictors of a positive genetic test in hypertrophic cardiomyopathy in the Brazilian population
title Clinical predictors of a positive genetic test in hypertrophic cardiomyopathy in the Brazilian population
title_full Clinical predictors of a positive genetic test in hypertrophic cardiomyopathy in the Brazilian population
title_fullStr Clinical predictors of a positive genetic test in hypertrophic cardiomyopathy in the Brazilian population
title_full_unstemmed Clinical predictors of a positive genetic test in hypertrophic cardiomyopathy in the Brazilian population
title_short Clinical predictors of a positive genetic test in hypertrophic cardiomyopathy in the Brazilian population
title_sort clinical predictors of a positive genetic test in hypertrophic cardiomyopathy in the brazilian population
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3995628/
https://www.ncbi.nlm.nih.gov/pubmed/24625281
http://dx.doi.org/10.1186/1471-2261-14-36
work_keys_str_mv AT marsigliajuliadahercarneiro clinicalpredictorsofapositivegenetictestinhypertrophiccardiomyopathyinthebrazilianpopulation
AT credidioflavialaghi clinicalpredictorsofapositivegenetictestinhypertrophiccardiomyopathyinthebrazilianpopulation
AT deoliveiratheogremenmimary clinicalpredictorsofapositivegenetictestinhypertrophiccardiomyopathyinthebrazilianpopulation
AT reisrafaelferreira clinicalpredictorsofapositivegenetictestinhypertrophiccardiomyopathyinthebrazilianpopulation
AT antunesmurillodeoliveira clinicalpredictorsofapositivegenetictestinhypertrophiccardiomyopathyinthebrazilianpopulation
AT dearaujoaloirqueiroz clinicalpredictorsofapositivegenetictestinhypertrophiccardiomyopathyinthebrazilianpopulation
AT pedrosarodrigopinto clinicalpredictorsofapositivegenetictestinhypertrophiccardiomyopathyinthebrazilianpopulation
AT barbosaferreirajoaomarcosbemfica clinicalpredictorsofapositivegenetictestinhypertrophiccardiomyopathyinthebrazilianpopulation
AT madycharles clinicalpredictorsofapositivegenetictestinhypertrophiccardiomyopathyinthebrazilianpopulation
AT kriegerjoseeduardo clinicalpredictorsofapositivegenetictestinhypertrophiccardiomyopathyinthebrazilianpopulation
AT arteagafernandezedmundo clinicalpredictorsofapositivegenetictestinhypertrophiccardiomyopathyinthebrazilianpopulation
AT pereiraalexandrecosta clinicalpredictorsofapositivegenetictestinhypertrophiccardiomyopathyinthebrazilianpopulation