Literature DB >> 17551851

Somatic alterations of the NF1 gene in an NF1 individual with multiple benign tumours (internal and external) and malignant tumour types.

Gill Spurlock1, Siân Griffiths, Jeremy Uff, Meena Upadhyaya.   

Abstract

Neurofibromatosis type 1 is a common familial cancer syndrome, affecting about 1 in every 4,000 individuals worldwide. We have carried out NF1 gene mutation analysis on DNA isolated from 25 tumours (dermal and plexiform neurofibromas, malignant peripheral nerve sheath tumour, MPNST), obtained at post-mortem from an NF1 patient. Macro and micro sequence alterations of the NF1 gene were studied by dHPLC, microsatellite, RFLP markers and multiplex ligation probe amplification (MLPA). The underlying germline mutation involves a deletion of exons 2 and 3. Of the 25 tumours studied from this patient, characterised somatic mutations were identified in 9 tumours, these were six small deletions (748del T, 2534-2557 del 24bp, 2843delA, 3047-3048 del GT, 4743del G, 7720-7721 delAA), an insertion 649 ins 73 bp, a non-sense mutation R1513X and a single splice site mutation, IVS4C-1 G>A, eight of these represent novel sequence changes in the gene. Evidence for loss of heterozygosity (LOH) was identified in DNA from 7 of the tumours. Each of the tumours analysed contained a different somatic NF1 mutation, indicating that each tumour is the result of an independent somatic event. The somatic mutation detection rate in this study is 64% (16/25), is one of the highest rates in genomic DNA reported so far in a single NF1 patient. Only 68 characterised NF1 somatic mutations have so far been reported and so our data will contribute to NF1 somatic mutational spectrum of the NF1 gene and will be important for understanding the molecular basis of NF1 tumorigenesis.

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Year:  2007        PMID: 17551851     DOI: 10.1007/s10689-007-9149-5

Source DB:  PubMed          Journal:  Fam Cancer        ISSN: 1389-9600            Impact factor:   2.375


  41 in total

1.  Somatic NF1 mutational spectrum in benign neurofibromas: mRNA splice defects are common among point mutations.

Authors:  E Serra; E Ars; A Ravella; A Sánchez; S Puig; T Rosenbaum; X Estivill; C Lázaro
Journal:  Hum Genet       Date:  2001-05       Impact factor: 4.132

2.  Von Recklinghausen neurofibromatosis. A clinical and population study in south-east Wales.

Authors:  S M Huson; P S Harper; D A Compston
Journal:  Brain       Date:  1988-12       Impact factor: 13.501

3.  Somatic loss of wild type NF1 allele in neurofibromas: Comparison of NF1 microdeletion and non-microdeletion patients.

Authors:  Thomas De Raedt; Ophélia Maertens; Magdalena Chmara; Hilde Brems; Ine Heyns; Raf Sciot; Elisa Majounie; Meena Upadhyaya; Sofie De Schepper; Frank Speleman; Ludwine Messiaen; Joris Robert Vermeesch; Eric Legius
Journal:  Genes Chromosomes Cancer       Date:  2006-10       Impact factor: 5.006

4.  Somatic mutations in the neurofibromatosis 1 gene in human tumors.

Authors:  Y Li; G Bollag; R Clark; J Stevens; L Conroy; D Fults; K Ward; E Friedman; W Samowitz; M Robertson
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5.  Toward a survey of somatic mutation of the NF1 gene in benign neurofibromas of patients with neurofibromatosis type 1.

Authors:  I Eisenbarth; K Beyer; W Krone; G Assum
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6.  Malignant peripheral nerve sheath tumours in neurofibromatosis 1.

Authors:  D G R Evans; M E Baser; J McGaughran; S Sharif; E Howard; A Moran
Journal:  J Med Genet       Date:  2002-05       Impact factor: 6.318

7.  Mutations affecting mRNA splicing are the most common molecular defects in patients with neurofibromatosis type 1.

Authors:  E Ars; E Serra; J García; H Kruyer; A Gaona; C Lázaro; X Estivill
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8.  Type 1 neurofibromatosis: a descriptive analysis of the disorder in 1,728 patients.

Authors:  J M Friedman; P H Birch
Journal:  Am J Med Genet       Date:  1997-05-16

9.  Somatic NF1 mutation spectra in a family with neurofibromatosis type 1: toward a theory of genetic modifiers.

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Journal:  Hum Mutat       Date:  2003-12       Impact factor: 4.878

10.  Neurofibromatosis type 1 gene as a mutational target in a mismatch repair-deficient cell type.

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  10 in total

1.  Analysis of NF1 somatic mutations in cutaneous neurofibromas from patients with high tumor burden.

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2.  Different sized somatic NF1 locus rearrangements in neurofibromatosis 1-associated malignant peripheral nerve sheath tumors.

Authors:  Eric Pasmant; Dominique Vidaud; Marcus Harrison; Meena Upadhyaya
Journal:  J Neurooncol       Date:  2010-08-05       Impact factor: 4.130

Review 3.  Neurofibromatosis type 1-associated tumours: their somatic mutational spectrum and pathogenesis.

Authors:  Sebastian Laycock-van Spyk; Nick Thomas; David N Cooper; Meena Upadhyaya
Journal:  Hum Genomics       Date:  2011-10       Impact factor: 4.639

4.  The spectrum of somatic and germline NF1 mutations in NF1 patients with spinal neurofibromas.

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Journal:  Neurogenetics       Date:  2009-02-17       Impact factor: 2.660

5.  Alphavirus production is inhibited in neurofibromin 1-deficient cells through activated RAS signalling.

Authors:  Olga A Kolokoltsova; Aaron M Domina; Andrey A Kolokoltsov; Robert A Davey; Scott C Weaver; Stanley J Watowich
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6.  Whole-exome sequencing of breast cancer, malignant peripheral nerve sheath tumor and neurofibroma from a patient with neurofibromatosis type 1.

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7.  Telomere erosion in NF1 tumorigenesis.

Authors:  Rhiannon E Jones; Julia W Grimstead; Ashni Sedani; Duncan Baird; Meena Upadhyaya
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8.  Whole Exome Sequencing of Growing and Non-Growing Cutaneous Neurofibromas from a Single Patient with Neurofibromatosis Type 1.

Authors:  Daniel L Faden; Saurabh Asthana; Tarik Tihan; Joseph DeRisi; Michel Kliot
Journal:  PLoS One       Date:  2017-01-18       Impact factor: 3.240

Review 9.  Do non-pathogenic variants of DNA mismatch repair genes modify neurofibroma load in neurofibromatosis type 1?

Authors:  Anja Harder
Journal:  Childs Nerv Syst       Date:  2022-01-08       Impact factor: 1.475

10.  Next-generation sequencing in the clinical genetic screening of patients with pheochromocytoma and paraganglioma.

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  10 in total

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