Fibrillin-1 gene analysis of Korean patients with spontaneous CSF hypovolemia

Headache. 2007 Jan;47(1):111-5. doi: 10.1111/j.1526-4610.2006.00635.x.

Abstract

Background: Mutations in different domains of the Fibrillin-1 (FBN1) gene may be responsible for the variable phenotypic expression of Marfan's syndrome that may present with CSF hypovolemia.

Objectives: To evaluate the association between mutations in the Fibrillin-1 (FBN1) gene and spontaneous CSF hypovolemia (SCH) in a Korean population.

Methods: We studied 10 consecutive patients with SCH without clinical characteristics of Marfan's syndrome. The genetic analysis was performed.

Results: Direct sequencing analysis of the FBN1 gene identified 15 genetic variations, of which 5 coding (3 synonymous, 2 nonsynonymous) and 8 intronic variations were listed in the single nucleotide polymorphism database (dbSNP). The other 2 variations, c.2728 - 12T > C in intron 21 and c.4582 - 19A > G in intron 35, were also observed in normal controls with estimated frequencies of 0.06 and 0.15, respectively.

Conclusions: We could not identify any FBN1 variations possibly associated with SCH in our study population.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Asian People / genetics*
  • Case-Control Studies
  • Cerebrospinal Fluid / physiology*
  • Cerebrospinal Fluid Pressure / genetics
  • Cerebrospinal Fluid Pressure / physiology
  • Female
  • Fibrillin-1
  • Fibrillins
  • Humans
  • Intracranial Hypotension / genetics*
  • Intracranial Hypotension / physiopathology
  • Korea
  • Male
  • Marfan Syndrome / genetics
  • Microfilament Proteins / genetics*
  • Middle Aged
  • Mutation / genetics
  • Phenotype
  • Polymorphism, Single Nucleotide / genetics

Substances

  • FBN1 protein, human
  • Fibrillin-1
  • Fibrillins
  • Microfilament Proteins