Literature DB >> 33443663

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

Filiz Hazan1, Semra Gürsoy2, Aycan Unalp3, Unsal Yılmaz3, Bengü Demirağ4, Sultan Aydin Köker5, Berk Ozyılmaz6, Kadri Murat Erdogan6, Önder Kalenderer7, Serkan Erkuş7, Müge Gürçınar8, Ajlan Tükün9.   

Abstract

Neurofibromatosis type 1 (NF1) is caused by mutations in the NF1 gene. This retrospective study aims to evaluate the clinical manifestations and brain magnetic resonance images (MRI) analysis in 60 genetically confirmed NF1 patients. The results of next-generation sequencing (NGS), Sanger sequencing, and MLPA of NF1 gene were evaluated. A total of 54 different variants were identified. Fourteen out of them were novel variants (25.9%). Patients who complied with NIH criteria had most frequently frameshift variants (11/32 patients), and those with only CALMs had missense variants (9/28 patients). Neurofibromatosis type 1 bright objects (NBOs) on T2-weighted MRI were detected in 42 patients (42/56; 75%). These brain lesions were detected mostly in basal ganglia and in cerebellar vermis. NBOs were detected more in the patients who complied with NIH criteria (80.6%) compared to those who were only CALMs (68%). While frameshift variants (33.3%) were the most common type variants in the patients who had NBOs, the most common variants were splicing (35.7%) and missense (35.7%) variants in the patients whose MRIs were normal. Frameshift variants (11/28 patients; 39.3%) were the most common in the patients with more than one brain locus involvement. Therefore, we consider that frameshift variants may be associated with increased incidence of NBOs and involvement of more than one brain locus. In addition, NBOs may occur less frequently in the patients with splicing variants. To our knowledge, this is the first study evaluated the relationship between NF1 gene variants and NBOs. Future studies may help us understand the etiology of NBOs.

Entities:  

Keywords:  Brain magnetic resonance images; Café-au-lait macules; NBOs; NF1 gene; Variants

Mesh:

Substances:

Year:  2021        PMID: 33443663     DOI: 10.1007/s10072-020-04988-0

Source DB:  PubMed          Journal:  Neurol Sci        ISSN: 1590-1874            Impact factor:   3.830


  43 in total

Review 1.  Café au lait spots: the pediatrician's perspective.

Authors:  M Tekin; J N Bodurtha; V M Riccardi
Journal:  Pediatr Rev       Date:  2001-03

Review 2.  Neurological manifestations of neurofibromatosis: a review.

Authors:  Michael Bayat; Allan Bayat
Journal:  Neurol Sci       Date:  2020-05-01       Impact factor: 3.307

3.  A major segment of the neurofibromatosis type 1 gene: cDNA sequence, genomic structure, and point mutations.

Authors:  R M Cawthon; R Weiss; G F Xu; D Viskochil; M Culver; J Stevens; M Robertson; D Dunn; R Gesteland; P O'Connell
Journal:  Cell       Date:  1990-07-13       Impact factor: 41.582

4.  Juvenile xanthogranuloma and nevus anemicus in the diagnosis of neurofibromatosis type 1.

Authors:  Faustine Ferrari; Alice Masurel; Laurence Olivier-Faivre; Pierre Vabres
Journal:  JAMA Dermatol       Date:  2014-01       Impact factor: 10.282

5.  The GAP-related domain of the neurofibromatosis type 1 gene product interacts with ras p21.

Authors:  G A Martin; D Viskochil; G Bollag; P C McCabe; W J Crosier; H Haubruck; L Conroy; R Clark; P O'Connell; R M Cawthon
Journal:  Cell       Date:  1990-11-16       Impact factor: 41.582

6.  Type 1 neurofibromatosis gene: identification of a large transcript disrupted in three NF1 patients.

Authors:  M R Wallace; D A Marchuk; L B Andersen; R Letcher; H M Odeh; A M Saulino; J W Fountain; A Brereton; J Nicholson; A L Mitchell
Journal:  Science       Date:  1990-07-13       Impact factor: 47.728

Review 7.  Neurofibromatosis type 1: a multidisciplinary approach to care.

Authors:  Angela C Hirbe; David H Gutmann
Journal:  Lancet Neurol       Date:  2014-08       Impact factor: 44.182

8.  Unidentified bright objects on brain MRI in children as a diagnostic criterion for neurofibromatosis type 1.

Authors:  José Roberto Lopes Ferraz Filho; Marcos Pontes Munis; Antonio Soares Souza; Rafael Angelo Sanches; Eni Maria Goloni-Bertollo; Erika Cristina Pavarino-Bertelli
Journal:  Pediatr Radiol       Date:  2008-01-30

9.  Predictive value of café au lait macules at initial consultation in the diagnosis of neurofibromatosis type 1.

Authors:  Kara S Nunley; Feng Gao; Anne C Albers; Susan J Bayliss; David H Gutmann
Journal:  Arch Dermatol       Date:  2009-08

Review 10.  Can the Cognitive Phenotype in Neurofibromatosis Type 1 (NF1) Be Explained by Neuroimaging? A Review.

Authors:  Eloïse Baudou; Federico Nemmi; Maëlle Biotteau; Stéphanie Maziero; Patrice Peran; Yves Chaix
Journal:  Front Neurol       Date:  2020-01-14       Impact factor: 4.003

View more
  1 in total

Review 1.  Image-Based Differentiation of Benign and Malignant Peripheral Nerve Sheath Tumors in Neurofibromatosis Type 1.

Authors:  Jun Liu; Jing-Ning Huang; Ming-Han Wang; Zhen-Yang Ni; Wei-Hao Jiang; Manhon Chung; Cheng-Jiang Wei; Zhi-Chao Wang
Journal:  Front Oncol       Date:  2022-05-23       Impact factor: 5.738

  1 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.