[Molecular genetic diagnosis of neurofibromatosis type I]

Orv Hetil. 2011 Mar 13;152(11):415-9. doi: 10.1556/OH.2011.29059.
[Article in Hungarian]

Abstract

Type 1 neurofibromatosis is an autosomal dominant hamartosis, caused by mutations of the gene neurofibromin-1. The variable clinical phenotype is characterized by café-au-lait spots, benign neurofibromas, axillary, inguinal hyperpigmentations, iris hamartomas, skeletal deformities and risk of neurofibroma-development. Pathogenic variations of neurofibromin-1 arise as de novo mutations in approx. 50% of the cases.

Aims: Molecular genetic testing of neurofibromin-1 gene has been performed in our department since 2008; the following report summarizes our experiences.

Methods: 40 patients, presenting symptoms of type 1 neurofibromatosis, were screened by sequencing or multiplex ligation-dependent probe amplification.

Results: Pathogenic alterations were identified in 31 cases, 8 patients presented novel mutations. In 8 affected, no mutations were detected by sequencing; one of these patients had a deletion affecting the entire gene.

Conclusions: Sequencing of the neurofibromin-1 gene and screening for rearrangements are useful in identifying pathogenic alterations in most of the cases.

Publication types

  • English Abstract

MeSH terms

  • Adult
  • Aged
  • Codon, Nonsense
  • Female
  • Gene Deletion
  • Genes, Neurofibromatosis 1*
  • Genetic Testing*
  • Humans
  • Male
  • Middle Aged
  • Mutagenesis, Insertional
  • Mutation*
  • Mutation, Missense
  • Neurofibromatosis 1 / diagnosis*
  • Neurofibromatosis 1 / genetics*
  • Neurofibromin 1 / genetics*
  • Pedigree
  • Sequence Analysis, DNA

Substances

  • Codon, Nonsense
  • Neurofibromin 1