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A Rare Case of Severe Congenital RYR1-Associated Myopathy

Congenital myopathies are a group of rare inherited diseases, defined by hypotonia and muscle weakness. We report clinical and genetic characteristics of a male preterm newborn, whose phenotype was characterized by severe hypotonia and hyporeactivity, serious respiratory distress syndrome that requi...

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Autores principales: Laforgia, Nicola, Capozza, Manuela, De Cosmo, Lucrezia, Di Mauro, Antonio, Baldassarre, Maria Elisabetta, Mercadante, Francesca, Torella, Anna Laura, Nigro, Vincenzo, Resta, Nicoletta
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Hindawi 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6092990/
https://www.ncbi.nlm.nih.gov/pubmed/30155320
http://dx.doi.org/10.1155/2018/6184185
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author Laforgia, Nicola
Capozza, Manuela
De Cosmo, Lucrezia
Di Mauro, Antonio
Baldassarre, Maria Elisabetta
Mercadante, Francesca
Torella, Anna Laura
Nigro, Vincenzo
Resta, Nicoletta
author_facet Laforgia, Nicola
Capozza, Manuela
De Cosmo, Lucrezia
Di Mauro, Antonio
Baldassarre, Maria Elisabetta
Mercadante, Francesca
Torella, Anna Laura
Nigro, Vincenzo
Resta, Nicoletta
author_sort Laforgia, Nicola
collection PubMed
description Congenital myopathies are a group of rare inherited diseases, defined by hypotonia and muscle weakness. We report clinical and genetic characteristics of a male preterm newborn, whose phenotype was characterized by severe hypotonia and hyporeactivity, serious respiratory distress syndrome that required mechanical ventilation, clubfoot, and other dysmorphic features. The diagnostic procedure was completed with the complete exome sequencing of the proband and of his parents and his sister, which showed new mutations in the ryanodine receptor gene (RYR1), which maps to chromosome 19q13.2 and encodes the skeletal muscle isoform of a calcium-release channel in the sarcoplasmic reticulum (RyR1). This report confirms that early diagnosis and accurate study of genomic disorders are very important, enabling proper genetic counselling of the reproductive risk, as well as disease prognosis and patient management.
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spelling pubmed-60929902018-08-28 A Rare Case of Severe Congenital RYR1-Associated Myopathy Laforgia, Nicola Capozza, Manuela De Cosmo, Lucrezia Di Mauro, Antonio Baldassarre, Maria Elisabetta Mercadante, Francesca Torella, Anna Laura Nigro, Vincenzo Resta, Nicoletta Case Rep Genet Case Report Congenital myopathies are a group of rare inherited diseases, defined by hypotonia and muscle weakness. We report clinical and genetic characteristics of a male preterm newborn, whose phenotype was characterized by severe hypotonia and hyporeactivity, serious respiratory distress syndrome that required mechanical ventilation, clubfoot, and other dysmorphic features. The diagnostic procedure was completed with the complete exome sequencing of the proband and of his parents and his sister, which showed new mutations in the ryanodine receptor gene (RYR1), which maps to chromosome 19q13.2 and encodes the skeletal muscle isoform of a calcium-release channel in the sarcoplasmic reticulum (RyR1). This report confirms that early diagnosis and accurate study of genomic disorders are very important, enabling proper genetic counselling of the reproductive risk, as well as disease prognosis and patient management. Hindawi 2018-08-01 /pmc/articles/PMC6092990/ /pubmed/30155320 http://dx.doi.org/10.1155/2018/6184185 Text en Copyright © 2018 Nicola Laforgia et al. https://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Laforgia, Nicola
Capozza, Manuela
De Cosmo, Lucrezia
Di Mauro, Antonio
Baldassarre, Maria Elisabetta
Mercadante, Francesca
Torella, Anna Laura
Nigro, Vincenzo
Resta, Nicoletta
A Rare Case of Severe Congenital RYR1-Associated Myopathy
title A Rare Case of Severe Congenital RYR1-Associated Myopathy
title_full A Rare Case of Severe Congenital RYR1-Associated Myopathy
title_fullStr A Rare Case of Severe Congenital RYR1-Associated Myopathy
title_full_unstemmed A Rare Case of Severe Congenital RYR1-Associated Myopathy
title_short A Rare Case of Severe Congenital RYR1-Associated Myopathy
title_sort rare case of severe congenital ryr1-associated myopathy
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6092990/
https://www.ncbi.nlm.nih.gov/pubmed/30155320
http://dx.doi.org/10.1155/2018/6184185
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