Literature DB >> 30308447

Phenotypic expression of a spectrum of Neurofibromatosis Type 1 (NF1) mutations identified through NGS and MLPA.

Maria Tsipi1, Myrto Poulou2, Irene Fylaktou2, Konstantina Kosma2, Eirini Tsoutsou2, Maria-Roser Pons3, Eleftheria Kokkinou3, Sofia Kitsiou-Tzeli4, Helen Fryssira2, Maria Tzetis2.   

Abstract

Neurofibromatosis Type 1 (NF1) is caused by mutations of the NF1 gene. The aim of this study was to identify the genetic causes underlying the disease, attempt possible phenotype/genotype correlations and add to the NF1 mutation spectrum. A screening protocol based on genomic DNA was established in 168 patients, encompassing sequencing of all coding exons and adjoining introns using a custom targeted next generation sequencing protocol and subsequent confirmation of findings with Sanger sequencing. MLPA was used to detect deletions/duplications and positive findings were confirmed by RNA analysis. All novel findings were evaluated according to ACMG Standards and guidelines for the interpretation of sequence variants with the aid of in-silico bioinformatic tools and family segregation analysis. A germline variant was identified in 145 patients (86%). In total 49 known and 70 novel variants in coding and non-coding regions were identified. Seven patients carried whole or partial gene deletions. NF1 patients, present with high phenotypic variability even in cases where the same germline disease causing variant has been identified. Our findings will contribute to a better knowledge of the genetic causes and the phenotypic expression related to the disease.
Copyright © 2018 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Café-au-lait macules; Disease causing variants; Genotyping; NF1; Neurofibromas; Next generation sequencing

Mesh:

Substances:

Year:  2018        PMID: 30308447     DOI: 10.1016/j.jns.2018.10.006

Source DB:  PubMed          Journal:  J Neurol Sci        ISSN: 0022-510X            Impact factor:   3.181


  10 in total

Review 1.  An update on the central nervous system manifestations of neurofibromatosis type 1.

Authors:  J Stephen Nix; Jaishri Blakeley; Fausto J Rodriguez
Journal:  Acta Neuropathol       Date:  2019-04-08       Impact factor: 17.088

2.  Deletion of the whole NF1 gene in a three-generation family with neurofibromatosis type 1.

Authors:  Qin Du; Hongxi Chen; Hongyu Zhou
Journal:  Neurol Sci       Date:  2021-06-05       Impact factor: 3.307

Review 3.  Diagnostic Pathology of Tumors of Peripheral Nerve.

Authors:  Sarra M Belakhoua; Fausto J Rodriguez
Journal:  Neurosurgery       Date:  2021-02-16       Impact factor: 4.654

4.  Evaluation of clinical findings and neurofibromatosis type 1 bright objects on brain magnetic resonance images of 60 Turkish patients with NF1 gene variants.

Authors:  Filiz Hazan; Semra Gürsoy; Aycan Unalp; Unsal Yılmaz; Bengü Demirağ; Sultan Aydin Köker; Berk Ozyılmaz; Kadri Murat Erdogan; Önder Kalenderer; Serkan Erkuş; Müge Gürçınar; Ajlan Tükün
Journal:  Neurol Sci       Date:  2021-01-14       Impact factor: 3.830

5.  Simultaneous Detection of NF1, SPRED1, LZTR1, and NF2 Gene Mutations by Targeted NGS in an Italian Cohort of Suspected NF1 Patients.

Authors:  Donatella Bianchessi; Maria Cristina Ibba; Veronica Saletti; Stefania Blasa; Tiziana Langella; Rosina Paterra; Giulia Anna Cagnoli; Giulia Melloni; Giulietta Scuvera; Federica Natacci; Claudia Cesaretti; Gaetano Finocchiaro; Marica Eoli
Journal:  Genes (Basel)       Date:  2020-06-19       Impact factor: 4.096

6.  Risk of Optic Pathway Glioma in Neurofibromatosis Type 1: No Evidence of Genotype-Phenotype Correlations in A Large Independent Cohort.

Authors:  Giulia Melloni; Marica Eoli; Claudia Cesaretti; Donatella Bianchessi; Maria Cristina Ibba; Silvia Esposito; Giulietta Scuvera; Guido Morcaldi; Roberto Micheli; Elena Piozzi; Sabrina Avignone; Luisa Chiapparini; Chiara Pantaleoni; Federica Natacci; Gaetano Finocchiaro; Veronica Saletti
Journal:  Cancers (Basel)       Date:  2019-11-21       Impact factor: 6.639

7.  Clinical Characteristics and Mutation Spectrum of Neurofibromatosis Type 1 in 27 Turkish Families.

Authors:  Shahrashoub Sharifi; Tuğba Kalaycı; Şükrü Palanduz; Şükrü Öztürk; Kıvanç Cefle
Journal:  Balkan Med J       Date:  2021-11       Impact factor: 2.021

8.  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

9.  [Genetic analysis and prenatal diagnosis of a sporadic family with neurofibromatosis type 1].

Authors:  Bei Liu; Yanmei Yang; Kai Yan; Min Chen; Liya Wang; Yingzhi Huang; Yeqing Qian; Minyue Dong
Journal:  Zhejiang Da Xue Xue Bao Yi Xue Ban       Date:  2019-06-25

10.  NF1 germline mutation in a Chinese family with colon cancer.

Authors:  Haizhou Lou; Chongya Zhai; Liu Gong; Hong Pan; Hongming Pan; Yihong Zhang; Mei Yang; Zimin Hu
Journal:  J Int Med Res       Date:  2020-08       Impact factor: 1.671

  10 in total

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