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Breakpoint characterization of a novel NF1 multiexonic deletion: a case showing expression of the mutated allele.

作者信息

Orzan Francesca, Stroppi Michela, Venturin Marco, Valero M Carmen, Hernández Concepcion, Riva Paola

机构信息

Department of Biology and Genetics, Medical Faculty, University of Milan, Milan, Italy.

出版信息

Neurogenetics. 2008 May;9(2):95-100. doi: 10.1007/s10048-007-0115-z. Epub 2008 Jan 10.

DOI:10.1007/s10048-007-0115-z
PMID:18196300
Abstract

Neurofibromatosis type 1 (NF1) is a common genetic disease caused by haploinsufficiency of the NF1 tumor-suppressor gene. Different pathogenetic mechanisms have been identified, with the majority (95%) causing intragenic lesions. Single or multiexon NF1 copy number changes occur in about 2% of patients, but little is known about the molecular mechanisms behind these intragenic deletions. We report here on the molecular characterization of a novel NF1 multiexonic deletion. The application of a multidisciplinary approach including multiplex ligation-dependent probe amplification, allelic segregation analysis, and fluorescent in situ hybridization allowed us to map the breakpoints in IVS27b and IVS48. Furthermore, the breakpoint junction was characterized by sequencing. Using bioinformatic analysis, we identified some recombinogenic motifs in close proximity to the centromeric and telomeric breakpoints and predicted the presence of a mutated messenger ribonucleic acid, which was deleted between exons 28 and 48 and encodes a neurofibromin that lacks some domains essential for its function. Through reverse transcriptase-polymerase chain reaction, the expression of the mutated allele was verified, showing the junction between exons 27b and 49 and, as expected, was not subjected to nonsense-mediated decay. Multiexonic deletions represent 2% of NF1 mutations, and until now, the breakpoint has been identified in only a few cases. The fine characterization of multiexonic deletions broadens the mutational repertoire of the NF1 gene, allowing for the identification of different pathogenetic mechanisms causing NF1.

摘要

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Syndecan-2 induces filopodia and dendritic spine formation via the neurofibromin-PKA-Ena/VASP pathway.Syndecan-2通过神经纤维瘤蛋白-PKA-Ena/VASP途径诱导丝状伪足和树突棘形成。
J Cell Biol. 2007 Jun 4;177(5):829-41. doi: 10.1083/jcb.200608121.
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The sec14 homology module of neurofibromin binds cellular glycerophospholipids: mass spectrometry and structure of a lipid complex.神经纤维瘤蛋白的sec14同源模块结合细胞甘油磷脂:脂质复合物的质谱分析与结构
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Reconstitution of the NF1 GAP-related domain in NF1-deficient human Schwann cells.
在缺乏NF1的人雪旺细胞中重建NF1 GAP相关结构域。
Biochem Biophys Res Commun. 2006 Sep 29;348(3):971-80. doi: 10.1016/j.bbrc.2006.07.159. Epub 2006 Aug 2.
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A novel bipartite phospholipid-binding module in the neurofibromatosis type 1 protein.1型神经纤维瘤病蛋白中一种新型的双组分磷脂结合模块。
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Spectrum of single- and multiexon NF1 copy number changes in a cohort of 1,100 unselected NF1 patients.1100例未经选择的1型神经纤维瘤病(NF1)患者队列中单个和多个外显子NF1拷贝数变化的谱系
Genes Chromosomes Cancer. 2006 Mar;45(3):265-76. doi: 10.1002/gcc.20289.
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Uncommon Alu-mediated NF1 microdeletion with a breakpoint inside the NF1 gene.罕见的由Alu介导的NF1微小缺失,断点位于NF1基因内部。
Genomics. 2005 Feb;85(2):273-9. doi: 10.1016/j.ygeno.2004.10.014.
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Novel mutations involving the NF1 gene coding sequence in neurofibromatosis type 1 patients from Taiwan.
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8
Translocation and gross deletion breakpoints in human inherited disease and cancer I: Nucleotide composition and recombination-associated motifs.人类遗传性疾病和癌症中的易位与大片段缺失断点I:核苷酸组成及与重组相关的基序
Hum Mutat. 2003 Sep;22(3):229-44. doi: 10.1002/humu.10254.
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Genotype analysis of the NF1 gene in the French Canadians from the Québec population.
Am J Med Genet. 2001 Dec 1;104(3):189-98.
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Identification and characterization of four novel large deletions in the human neurofibromatosis type 1 (NF1) gene.人类1型神经纤维瘤病(NF1)基因中四个新型大片段缺失的鉴定与特征分析。
Hum Mutat. 2001 Dec;18(6):549-50. doi: 10.1002/humu.1241.