Progressive late-onset sensorineural hearing loss and vestibular impairment with vertigo (DFNA9/COCH): longitudinal analyses in a belgian family

Otol Neurotol. 2003 Sep;24(5):743-8. doi: 10.1097/00129492-200309000-00009.

Abstract

Objective: To evaluate audiometric and vestibular signs and symptoms in a new DFNA9 family.

Setting: Tertiary referral centers.

Methods: A multigeneration Belgian family with late-onset progressive sensorineural hearing loss and concomitant ves-tibular impairment with an autosomal dominant pattern of inheritance underwent clinical and genetic evaluation. Medical history was recorded. Blood samples were taken for genetic linkage and mutation analyses. Pure-tone audiometry, speech audiometry and vestibular examinations were performed. Onset and progression in hearing impairment were evaluated with linear regression analysis of longitudinal threshold-on-age data.

Results: Linkage to DFNA9 was confirmed and mutation analysis revealed a P51S mutation in the COCH gene. Several patients had a Ménière's-like presentation. All patients developed late-onset progressive sensorineural hearing loss eventually leading to severe deafness and vestibular failure.

MeSH terms

  • Adult
  • Aged
  • Aged, 80 and over
  • Audiometry, Pure-Tone
  • Belgium
  • Chromosome Aberrations
  • Chromosome Mapping
  • DNA Mutational Analysis
  • Deafness / diagnosis
  • Deafness / genetics*
  • Disease Progression
  • Extracellular Matrix Proteins
  • Female
  • Genes, Dominant
  • Genetic Carrier Screening
  • Hearing Loss, Sensorineural / diagnosis
  • Hearing Loss, Sensorineural / genetics*
  • Humans
  • Karyotyping
  • Longitudinal Studies
  • Male
  • Meniere Disease / diagnosis
  • Meniere Disease / genetics*
  • Middle Aged
  • Presbycusis / diagnosis
  • Presbycusis / genetics*
  • Proteins / genetics*
  • Speech Reception Threshold Test
  • Vestibular Diseases / diagnosis
  • Vestibular Diseases / genetics*
  • Vestibular Function Tests

Substances

  • COCH protein, human
  • Extracellular Matrix Proteins
  • Proteins