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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...
Autores principales: | , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2021
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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. |
format | Online Article Text |
id | pubmed-8147995 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2021 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
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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