Logo image
Performance of Cochlear Implant Recipients With GJB2-Related Deafness
Journal article   Open access   Peer reviewed

Performance of Cochlear Implant Recipients With GJB2-Related Deafness

Glenn E Green, Daryl A Scott, Joshua M McDonald, Holly F.B Teagle, Bruce J Tomblin, Linda J Spencer, George G Woodworth, John F Knutson, Bruce J Gantz, Val C Sheffield, …
American journal of medical genetics, Vol.109(3), pp.167-170
05/01/2002
DOI: 10.1002/ajmg.10330
PMCID: PMC3212411
PMID: 11977173
url
https://doi.org/10.1002/ajmg.10330View
Published (Version of record) Open Access

Abstract

Congenital profound hearing loss affects 0.05–0.1% of children and has many causes, some of which are associated with cognitive delay. For prelingually-deafened cochlear implant recipients, the etiology of deafness is usually unknown. Mutations in GJB2 have been established as the most common cause of heritable deafness in the United States. In this report, we identify cochlear implant recipients with GJB2 -related deafness and examine the performance of these individuals. Cochlear implant recipients received a battery of perceptive, cognitive, and reading tests. Neither subjects nor examiners knew the etiology of deafness in these individuals. The implant recipients were then examined for mutations in GJB2 using an allele-specific polymerase chain reaction assay, single-strand conformation polymorphism analysis, and direct sequencing. GJB2 mutations were the leading cause of congenital deafness among the cochlear implant recipients screened. Cochlear implant recipients with GJB2 -related deafness read within one standard deviation of hearing controls better than other congenitally deaf cochlear implant recipients and non-cochlear implant recipients. Individuals with congenital deafness should be offered GJB2 screening. Positive results establish an etiologic diagnosis and provide prognostic, genetic, and therapeutic information. Effective rehabilitation for profoundly deaf individuals with GJB2 -related deafness is possible through cochlear implantation.
GJB2 deafness reading performance cochlear implantation connexin 26

Details

Metrics

Logo image