New Mutations in NEB Gene Discovered by Targeted Next-Generation Sequencing in Nemaline Myopathy Italian Patients

被引:15
作者
Piga, Daniela [1 ]
Magri, Francesca [1 ]
Ronchi, Dario [1 ]
Corti, Stefania [1 ]
Cassandrini, Denise [2 ]
Mercuri, Eugenio [3 ]
Tasca, Giorgio [4 ]
Bertini, Enrico [5 ]
Fattori, Fabiana [5 ]
Toscano, Antonio [6 ]
Messina, Sonia [6 ]
Moroni, Isabella [7 ,8 ,9 ]
Mora, Marina [7 ,8 ,9 ]
Moggio, Maurizio [10 ]
Colombo, Irene [10 ]
Giugliano, Teresa [11 ,12 ]
Pane, Marika [3 ]
Fiorillo, Chiara [13 ]
D'Amico, Adele [5 ]
Bruno, Claudio [13 ]
Nigro, Vincenzo [11 ,12 ]
Bresolin, Nereo [1 ]
Comi, Giacomo Pietro [1 ]
机构
[1] Univ Milan, Dept Pathophysiol & Transplantat DEPT, IRCCS Fdn Ca Granda, Dino Ferrari Ctr,Neurol Unit,Osped Maggiore Polic, Via F Sforza 35, I-20122 Milan, Italy
[2] IRCCS Stella Maris, Mol Med Unit, Pisa, Italy
[3] Univ Cattolica Sacro Cuore, Dept Pediat, Policlin Gemelli, Rome, Italy
[4] Don Carlo Gnocchi Onlus Fdn, Milan, Italy
[5] Bambino Gesu Pediat Hosp, Dept Labs, Unit Mol Med Neuromuscular & Neurodegenerat Disor, Rome, Italy
[6] Univ Messina, Dept Neurosci Psychiat & Anesthesiol, AOU Policlin G Martino, Messina, Italy
[7] Fdn Carlo Besta IRCCS Neurol Inst, Pediat Unit, Milan, Italy
[8] Fdn Carlo Besta IRCCS Neurol Inst, Neuromuscular Dis Unit, Milan, Italy
[9] Fdn Carlo Besta IRCCS Neurol Inst, Neuroimmunol Unit, Milan, Italy
[10] Univ Milan, Dino Ferrari Ctr, Neuromuscular Unit, IRCCS Fdn Ca Granda Osped Maggiore Policlin, Milan, Italy
[11] Univ Naples 2, Dept Biochem Biophys & Gen Pathol, Naples, Italy
[12] Telethon Inst Genet & Med, Naples, Italy
[13] Giannina Gaslini Inst, Ctr Myol & Neurodegenerat Dis, Genoa, Italy
关键词
NEB mutations; Nemaline myopathy; Next-generation sequencing; COMPARATIVE GENOMIC HYBRIDIZATION; MUSCLE ALPHA-ACTIN; NEBULIN GENE; CONGENITAL MYOPATHY; DISTAL MYOPATHY; IDENTIFICATION; CORES; RODS; UPDATE; TPM2;
D O I
10.1007/s12031-016-0739-2
中图分类号
Q5 [生物化学]; Q7 [分子生物学];
学科分类号
071010 ; 081704 ;
摘要
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.
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收藏
页码:351 / 359
页数:9
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