TY - JOUR
T1 - New Mutations in NEB Gene Discovered by Targeted Next-Generation Sequencing in Nemaline Myopathy Italian Patients
AU - Piga, Daniela
AU - Magri, Francesca
AU - Ronchi, Dario
AU - Corti, Stefania
AU - Cassandrini, Denise
AU - Mercuri, Eugenio Maria
AU - Tasca, Giorgio
AU - Bertini, Enrico
AU - Bertini, Enrico Silvio
AU - Fattori, Fabiana
AU - Toscano, Antonio
AU - Messina, Sonia
AU - Moroni, Isabella
AU - Mora, Marina
AU - Moggio, Maurizio
AU - Colombo, Irene
AU - Giugliano, Teresa
AU - Pane, Marika
AU - Fiorillo, Chiara
AU - D’Amico, Adele
AU - D'Amico, Adele
AU - Bruno, Claudio
AU - Nigro, Vincenzo
AU - Bresolin, Nereo
AU - Comi, Giacomo Pietro
PY - 2016
Y1 - 2016
N2 - Nemaline myopathy represents a group of clinically and genetically heterogeneous neuromuscular disorders. Different clinical-genetic entities have been characterized in the last few years, with implications for diagnostics and genetic counseling. Fifty percent of nemaline myopathy forms are due to NEB mutations, but genetic analysis of this large and complex gene by Sanger sequencing is time consuming and expensive. We selected 10 Italian patients with clinical and biopsy features suggestive for nemaline myopathy and negative for ACTA1, TPM2 and TPM3 mutations. We applied a targeted next-generation sequencing strategy designed to analyse NEB coding regions, the relative full introns and the promoter. We also evaluated copy number variations (by CGH array) and transcriptional changes by RNA Sanger sequencing, whenever possible. This combined strategy revealed 11 likely pathogenic variants in 8 of 10 patients. The molecular diagnosis was fully achieved in 3 of 8 patients, while only one heterozygous mutation was observed in 5 subjects. This approach revealed to be a fast and cost-effective way to analyse the large NEB gene in a small group of patients and might be promising for the detection of pathological variants of other genes featuring large coding regions and lacking mutational hotspots.
AB - Nemaline myopathy represents a group of clinically and genetically heterogeneous neuromuscular disorders. Different clinical-genetic entities have been characterized in the last few years, with implications for diagnostics and genetic counseling. Fifty percent of nemaline myopathy forms are due to NEB mutations, but genetic analysis of this large and complex gene by Sanger sequencing is time consuming and expensive. We selected 10 Italian patients with clinical and biopsy features suggestive for nemaline myopathy and negative for ACTA1, TPM2 and TPM3 mutations. We applied a targeted next-generation sequencing strategy designed to analyse NEB coding regions, the relative full introns and the promoter. We also evaluated copy number variations (by CGH array) and transcriptional changes by RNA Sanger sequencing, whenever possible. This combined strategy revealed 11 likely pathogenic variants in 8 of 10 patients. The molecular diagnosis was fully achieved in 3 of 8 patients, while only one heterozygous mutation was observed in 5 subjects. This approach revealed to be a fast and cost-effective way to analyse the large NEB gene in a small group of patients and might be promising for the detection of pathological variants of other genes featuring large coding regions and lacking mutational hotspots.
KW - Cellular and Molecular Neuroscience
KW - Medicine (all)
KW - NEB mutations
KW - Nemaline myopathy
KW - Next-generation sequencing
KW - Cellular and Molecular Neuroscience
KW - Medicine (all)
KW - NEB mutations
KW - Nemaline myopathy
KW - Next-generation sequencing
UR - http://hdl.handle.net/10807/92049
UR - http://www.springer.com/humana+press/journal/12031
U2 - 10.1007/s12031-016-0739-2
DO - 10.1007/s12031-016-0739-2
M3 - Article
SN - 0895-8696
VL - 59
SP - 351
EP - 359
JO - Journal of Molecular Neuroscience
JF - Journal of Molecular Neuroscience
ER -