Literature DB >> 27074763

Identification and characterization of NF1 splicing mutations in Korean patients with neurofibromatosis type 1.

Mi-Ae Jang1, Young-Eun Kim2, Sun Kyung Kim3, Myoung-Keun Lee3, Jong-Won Kim3, Chang-Seok Ki3.   

Abstract

Neurofibromatosis type I (NF1) is an autosomal dominant genetic disorder caused by NF1 mutations. Although mutations affecting mRNA splicing are the most common molecular defects in NF1, few studies have analyzed genomic DNA (gDNA)-mRNA correlations in Korean NF1 patients. In this study, we investigated 28 unrelated NF1 patients who showed splicing alterations in reverse transcription-PCR of NF1 mRNA and identified 24 different NF1 splicing mutations, 9 of which were novel. These mutations can be categorized into five groups: exon skipping resulting from mutations at authentic 5' and 3' splice sites (type I, 46%), cryptic exon inclusion caused by deep intronic mutations (type II, 8%), creation of new splice sites causing loss of exonic sequences (type III, 8%), activation of cryptic splice sites due to disruption of authentic splice sites (type IV, 25%) and exonic sequence alterations causing exon skipping (type V, 13%). In total, 42% of all splicing mutations did not involve the conserved AG/GT dinucleotides of the splice sites, making it difficult to identify the correct mutation sites at the gDNA level. These results add to the mutational spectrum of NF1 and further elucidate the gDNA-mRNA correlations of NF1 mutations.

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Year:  2016        PMID: 27074763     DOI: 10.1038/jhg.2016.33

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  21 in total

1.  Characterisation of a novel splice donor mutation affecting position +1 in intron 18 of the NF-1 gene.

Authors:  S M Purandare; W G Lanyon; R Arngrimsson; J M Connor
Journal:  Hum Mol Genet       Date:  1995-04       Impact factor: 6.150

2.  A highly sensitive genetic protocol to detect NF1 mutations.

Authors:  María Carmen Valero; Yolanda Martín; Elisabete Hernández-Imaz; Alba Marina Hernández; Germán Meleán; Ana María Valero; Francisco Javier Rodríguez-Álvarez; Dolores Tellería; Concepción Hernández-Chico
Journal:  J Mol Diagn       Date:  2011-03       Impact factor: 5.568

3.  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
Journal:  Am J Hum Genet       Date:  2000-02       Impact factor: 11.025

4.  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
Journal:  Hum Mol Genet       Date:  2000-01-22       Impact factor: 6.150

5.  Extensive in silico analysis of NF1 splicing defects uncovers determinants for splicing outcome upon 5' splice-site disruption.

Authors:  K Wimmer; X Roca; H Beiglböck; T Callens; J Etzler; A R Rao; A R Krainer; C Fonatsch; L Messiaen
Journal:  Hum Mutat       Date:  2007-06       Impact factor: 4.878

6.  Neurofibromatosis type 1: a single center's experience in Korea.

Authors:  Min Jeong Kim; Chong Kun Cheon
Journal:  Korean J Pediatr       Date:  2014-09-30

7.  NF1 molecular characterization and neurofibromatosis type I genotype-phenotype correlation: the French experience.

Authors:  Audrey Sabbagh; Eric Pasmant; Apolline Imbard; Armelle Luscan; Magali Soares; Hélène Blanché; Ingrid Laurendeau; Salah Ferkal; Michel Vidaud; Stéphane Pinson; Christine Bellanné-Chantelot; Dominique Vidaud; Béatrice Parfait; Pierre Wolkenstein
Journal:  Hum Mutat       Date:  2013-08-26       Impact factor: 4.878

8.  Two recurrent nonsense mutations and a 4 bp deletion in a quasi-symmetric element in exon 37 of the NF1 gene.

Authors:  P N Robinson; A Böddrich; H Peters; S Tinschert; A Buske; D Kaufmann; P Nürnberg
Journal:  Hum Genet       Date:  1995-07       Impact factor: 4.132

9.  Mutation spectrum of NF1 and clinical characteristics in 78 Korean patients with neurofibromatosis type 1.

Authors:  Jung Min Ko; Young Bae Sohn; Seon Yong Jeong; Hyon-Ju Kim; Ludwine M Messiaen
Journal:  Pediatr Neurol       Date:  2013-06       Impact factor: 3.372

10.  Fifty-four novel mutations in the NF1 gene and integrated analyses of the mutations that modulate splicing.

Authors:  Weihong Xu; Xiao Yang; Xiaoxia Hu; Shibo Li
Journal:  Int J Mol Med       Date:  2014-04-24       Impact factor: 4.101

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  5 in total

1.  Hereditary cancer genes are highly susceptible to splicing mutations.

Authors:  Christy L Rhine; Kamil J Cygan; Rachel Soemedi; Samantha Maguire; Michael F Murray; Sean F Monaghan; William G Fairbrother
Journal:  PLoS Genet       Date:  2018-03-05       Impact factor: 5.917

2.  Analysis of Pathogenic Pseudoexons Reveals Novel Mechanisms Driving Cryptic Splicing.

Authors:  Niall P Keegan; Steve D Wilton; Sue Fletcher
Journal:  Front Genet       Date:  2022-01-24       Impact factor: 4.772

3.  Genotype-Phenotype Correlations in Neurofibromatosis Type 1: Identification of Novel and Recurrent NF1 Gene Variants and Correlations with Neurocognitive Phenotype.

Authors:  Filomena Napolitano; Milena Dell'Aquila; Chiara Terracciano; Giuseppina Franzese; Maria Teresa Gentile; Giulio Piluso; Claudia Santoro; Davide Colavito; Anna Patanè; Paolo De Blasiis; Simone Sampaolo; Simona Paladino; Mariarosa Anna Beatrice Melone
Journal:  Genes (Basel)       Date:  2022-06-23       Impact factor: 4.141

4.  Two Novel NF1 Pathogenic Variants Causing the Creation of a New Splice Site in Patients With Neurofibromatosis Type I.

Authors:  Vita Setrajcic Dragos; Ana Blatnik; Gasper Klancar; Vida Stegel; Mateja Krajc; Olga Blatnik; Srdjan Novakovic
Journal:  Front Genet       Date:  2019-08-22       Impact factor: 4.599

5.  Performance Evaluation of SpliceAI for the Prediction of Splicing of NF1 Variants.

Authors:  Changhee Ha; Jong-Won Kim; Ja-Hyun Jang
Journal:  Genes (Basel)       Date:  2021-08-25       Impact factor: 4.096

  5 in total

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