Audiological findings in 100 USH2 patients

Clin Genet. 2012 Nov;82(5):433-8. doi: 10.1111/j.1399-0004.2011.01772.x. Epub 2011 Sep 30.

Abstract

Bilateral sensorineural hearing loss (HL), classically described as mild to severe with a typically down-sloping audiometric configuration, is the earliest symptom occurring in Usher syndrome type II (USH2). Audiological findings were analyzed in a total of 100 USH2 patients (92 families) divided into three groups according to the gene involved: 88 USH2A, 10 GPR98 and 2 DFNB31 patients. A fine analysis of audiograms was performed (pure tone average, degree of severity, configuration). The median age of HL diagnosis was 5 years (range 8 months-31 years) although the median age at USH2 diagnosis was 34.5 (range 8-76). Moderate HL was predominant (76%) and a gently down-sloping configuration characterized most audiograms (66%). No statistically significant difference was found between USH2A and GPR98 patients but a tendency was clearly noted for more GPR98 patients to present with severe hearing loss. It is not possible to predict the mutated gene from audiograms.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Audiology / methods*
  • Audiometry / methods
  • Child
  • Child, Preschool
  • Cross-Sectional Studies
  • Extracellular Matrix Proteins / genetics*
  • Female
  • Genotype
  • Hearing Loss, Sensorineural / diagnosis*
  • Hearing Loss, Sensorineural / genetics
  • Humans
  • Infant
  • Male
  • Membrane Proteins / genetics
  • Mutation
  • Receptors, G-Protein-Coupled / genetics
  • Young Adult

Substances

  • ADGRV1 protein, human
  • Extracellular Matrix Proteins
  • Membrane Proteins
  • Receptors, G-Protein-Coupled
  • USH2A protein, human
  • WHRN protein, human