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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...
Autores principales: | , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Hindawi
2018
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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. |
format | Online Article Text |
id | pubmed-6092990 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2018 |
publisher | Hindawi |
record_format | MEDLINE/PubMed |
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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