Literature DB >> 12082509

A patient severely affected by spinal neurofibromas carries a recurrent splice site mutation in the NF1 gene.

Katharina Wimmer1, Manfred Mühlbauer, Markus Eckart, Tom Callens, Helga Rehder, Thomas Birkner, Jules G Leroy, Christa Fonatsch, Ludwine Messiaen.   

Abstract

Spinal neurofibromas are found in up to 38% of NF1 patients. However, they cause clinical implications only in about 5% of the patients. In contrast, multiple symptomatic spinal neurofibromas are the main clinical finding in patients with familial spinal neurofibromatosis. Familial spinal neurofibromatosis has been considered to be a distinct clinical form of neurofibromatosis. Linkage analysis in two families and identification of a NF1 gene mutation in a third family strongly associate spinal neurofibromatosis with the NF1 gene. We describe a NF1 patient who satisfies the NIH diagnostic criteria and has severe spinal involvement with bilateral spinal root neurofibromas at every level. A recurrent splice site mutation (IVS19b-3C>G) was identified in the NF1 gene in the patient. We discuss the possibility that the clinical picture of this patient represents an additional example of spinal neurofibromatosis. By comparison of the clinical expression of NF1 in this patient and that in another patient with the identical mutation the hypothesis that spinal neurofibromatosis is associated with a particular mutation is highly unlikely. The involvement of other genes linked to the NF1 gene or modifying genes is currently the most likely explanation for the clinical phenotype of spinal neurofibromatosis.

Entities:  

Mesh:

Substances:

Year:  2002        PMID: 12082509     DOI: 10.1038/sj.ejhg.5200807

Source DB:  PubMed          Journal:  Eur J Hum Genet        ISSN: 1018-4813            Impact factor:   4.246


  6 in total

1.  Familial spinal neurofibromatosis due to a multiexonic NF1 gene deletion.

Authors:  Antonio Pizzuti; Irene Bottillo; Francesca Inzana; Valentina Lanari; Francesca Buttarelli; Isabella Torrente; Anna Teresa Giallonardo; Alessandro De Luca; Bruno Dallapiccola
Journal:  Neurogenetics       Date:  2011-03-02       Impact factor: 2.660

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.  Spinal neurofibromatosis associated with classical neurofibromatosis type 1: genetic characterisation of an atypical case.

Authors:  Kursat bora Carman; Ayten Yakut; Banu Anlar; Sukriye Ayter
Journal:  BMJ Case Rep       Date:  2013-02-14

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

Authors:  Meena Upadhyaya; Gill Spurlock; Lan Kluwe; Nadia Chuzhanova; Emma Bennett; Nick Thomas; Abhijit Guha; Victor Mautner
Journal:  Neurogenetics       Date:  2009-02-17       Impact factor: 2.660

Review 5.  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

6.  Can the diagnosis of NF1 be excluded clinically? A lack of pigmentary findings in families with spinal neurofibromatosis demonstrates a limitation of clinical diagnosis.

Authors:  Emma Mm Burkitt Wright; Emma Sach; Saba Sharif; Oliver Quarrell; Thomas Carroll; Richard W Whitehouse; Meena Upadhyaya; Susan M Huson; D Gareth R Evans
Journal:  J Med Genet       Date:  2013-06-28       Impact factor: 6.318

  6 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.