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Role of RNA in Molecular Diagnosis of MADD Patients

The electron-transfer flavoprotein dehydrogenase gene (ETFDH) encodes the ETF-ubiquinone oxidoreductase (ETF-QO) and has been reported to be the major cause of multiple acyl-CoA dehydrogenase deficiency (MADD). In this study, we present the clinical and molecular diagnostic challenges, at the DNA an...

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Autores principales: Nogueira, Célia, Silva, Lisbeth, Marcão, Ana, Sousa, Carmen, Fonseca, Helena, Rocha, Hugo, Campos, Teresa, Teles, Elisa Leão, Rodrigues, Esmeralda, Janeiro, Patrícia, Gaspar, Ana, Vilarinho, Laura
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8147995/
https://www.ncbi.nlm.nih.gov/pubmed/34064479
http://dx.doi.org/10.3390/biomedicines9050507
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author Nogueira, Célia
Silva, Lisbeth
Marcão, Ana
Sousa, Carmen
Fonseca, Helena
Rocha, Hugo
Campos, Teresa
Teles, Elisa Leão
Rodrigues, Esmeralda
Janeiro, Patrícia
Gaspar, Ana
Vilarinho, Laura
author_facet Nogueira, Célia
Silva, Lisbeth
Marcão, Ana
Sousa, Carmen
Fonseca, Helena
Rocha, Hugo
Campos, Teresa
Teles, Elisa Leão
Rodrigues, Esmeralda
Janeiro, Patrícia
Gaspar, Ana
Vilarinho, Laura
author_sort Nogueira, Célia
collection PubMed
description The electron-transfer flavoprotein dehydrogenase gene (ETFDH) encodes the ETF-ubiquinone oxidoreductase (ETF-QO) and has been reported to be the major cause of multiple acyl-CoA dehydrogenase deficiency (MADD). In this study, we present the clinical and molecular diagnostic challenges, at the DNA and RNA levels, involved in establishing the genotype of four MADD patients with novel ETFDH variants: a missense variant, two deep intronic variants and a gross deletion. RNA sequencing allowed the identification of the second causative allele in all studied patients. Simultaneous DNA and RNA investigation can increase the number of MADD patients that can be confirmed following the suggestive data results of an expanded newborn screening program. In clinical practice, accurate identification of pathogenic mutations is fundamental, particularly with regard to diagnostic, prognostic, therapeutic and ethical issues. Our study highlights the importance of RNA studies for a definitive molecular diagnosis of MADD patients, expands the background of ETFDH mutations and will be important in providing an accurate genetic counseling and a prenatal diagnosis for the affected families.
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spelling pubmed-81479952021-05-26 Role of RNA in Molecular Diagnosis of MADD Patients Nogueira, Célia Silva, Lisbeth Marcão, Ana Sousa, Carmen Fonseca, Helena Rocha, Hugo Campos, Teresa Teles, Elisa Leão Rodrigues, Esmeralda Janeiro, Patrícia Gaspar, Ana Vilarinho, Laura Biomedicines Case Report The electron-transfer flavoprotein dehydrogenase gene (ETFDH) encodes the ETF-ubiquinone oxidoreductase (ETF-QO) and has been reported to be the major cause of multiple acyl-CoA dehydrogenase deficiency (MADD). In this study, we present the clinical and molecular diagnostic challenges, at the DNA and RNA levels, involved in establishing the genotype of four MADD patients with novel ETFDH variants: a missense variant, two deep intronic variants and a gross deletion. RNA sequencing allowed the identification of the second causative allele in all studied patients. Simultaneous DNA and RNA investigation can increase the number of MADD patients that can be confirmed following the suggestive data results of an expanded newborn screening program. In clinical practice, accurate identification of pathogenic mutations is fundamental, particularly with regard to diagnostic, prognostic, therapeutic and ethical issues. Our study highlights the importance of RNA studies for a definitive molecular diagnosis of MADD patients, expands the background of ETFDH mutations and will be important in providing an accurate genetic counseling and a prenatal diagnosis for the affected families. MDPI 2021-05-04 /pmc/articles/PMC8147995/ /pubmed/34064479 http://dx.doi.org/10.3390/biomedicines9050507 Text en © 2021 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 Case Report
Nogueira, Célia
Silva, Lisbeth
Marcão, Ana
Sousa, Carmen
Fonseca, Helena
Rocha, Hugo
Campos, Teresa
Teles, Elisa Leão
Rodrigues, Esmeralda
Janeiro, Patrícia
Gaspar, Ana
Vilarinho, Laura
Role of RNA in Molecular Diagnosis of MADD Patients
title Role of RNA in Molecular Diagnosis of MADD Patients
title_full Role of RNA in Molecular Diagnosis of MADD Patients
title_fullStr Role of RNA in Molecular Diagnosis of MADD Patients
title_full_unstemmed Role of RNA in Molecular Diagnosis of MADD Patients
title_short Role of RNA in Molecular Diagnosis of MADD Patients
title_sort role of rna in molecular diagnosis of madd patients
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8147995/
https://www.ncbi.nlm.nih.gov/pubmed/34064479
http://dx.doi.org/10.3390/biomedicines9050507
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