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Titolo:
Neuroepithelial defects of the inner ear in a new allele of the mouse mutation Ames waltzer
Autore:
Alagramam, KN; Zahorsky-Reeves, J; Wright, CG; Pawlowski, KS; Erway, LC; Stubbs, L; Woychik, RP;
Indirizzi:
Case Western Reserve Univ, Rainbow Babies & Childrens Hosp, Dept Pediat, Cleveland, OH 44106 USA Case Western Reserve Univ Cleveland OH USA 44106 Cleveland, OH 44106 USA Univ Tennessee, Dept Pathol, Knoxville, TN 37996 USA Univ Tennessee Knoxville TN USA 37996 ept Pathol, Knoxville, TN 37996 USA Univ Texas, SW Med Ctr, Dept Otorhinolaryngol, Dallas, TX 75235 USA Univ Texas Dallas TX USA 75235 ept Otorhinolaryngol, Dallas, TX 75235 USA Univ Cincinnati, Dept Biol Sci, Cincinnati, OH 45221 USA Univ Cincinnati Cincinnati OH USA 45221 iol Sci, Cincinnati, OH 45221 USA Univ Calif Lawrence Livermore Natl Lab, Ctr Human Genome, Livermore, CA 94550 USA Univ Calif Lawrence Livermore Natl Lab Livermore CA USA 94550 A 94550 USA
Titolo Testata:
HEARING RESEARCH
fascicolo: 1-2, volume: 148, anno: 2000,
pagine: 181 - 191
SICI:
0378-5955(200010)148:1-2<181:NDOTIE>2.0.ZU;2-R
Fonte:
ISI
Lingua:
ENG
Soggetto:
HEREDITARY HEARING-LOSS; COCHLEAR HAIR-CELLS; INSERTIONAL MUTATION; MICE; DEAFNESS; GENES; MOUSE-CHROMOSOME-10; STRAINS;
Keywords:
inner ear; mouse mutant; Ames waltzer;
Tipo documento:
Article
Natura:
Periodico
Settore Disciplinare:
Life Sciences
Citazioni:
28
Recensione:
Indirizzi per estratti:
Indirizzo: Woychik, RP Pfizer Global Res & Dev, Alameda Labs, 1501 Harbor Bay Pkwy, Alameda, CA 94502 USA Pfizer Global Res & Dev 1501 Harbor Bay Pkwy Alameda CA USA 94502
Citazione:
K.N. Alagramam et al., "Neuroepithelial defects of the inner ear in a new allele of the mouse mutation Ames waltzer", HEARING RES, 148(1-2), 2000, pp. 181-191

Abstract

This report presents new findings regarding a recessive insertional mutation in the transgenic line TgN2742Rpw that causes deafness and circling behavior in mice homozygous for the mutation. The mutant locus was mapped to a region on mouse chromosome 10 close to three spontaneous recessive mutations causing deafness: Ames waltzer (av), Waltzer (v), and Jackson circler (jc) Complementation testing revealed that the TgN2742Rpw mutation is allelic with av. Histological and auditory brainstem response (ABR) evaluation of animals that have the new allele balanced with the av(J) allele (called compound heterozygotes, TgN2742Rpw/av(J)) supports our genetic analysis. ABR evaluation shows complete absence of auditory response throughout the life span of TgN2742Rpw/av(J) compound heterozygotes. Scanning electron microscopyrevealed abnormalities of inner and outer hair cell stereocilia in the cochleae of TgN2742Rpw mutants at 10 days after bi;th (DAB). The organ of Corti subsequently undergoes degeneration, leading to nearly complete loss of the cochlear neuroepithelium in older mutants by about 50 DAB. The vestibular neuroepithelia remain morphologically normal until at least 30 DAB. However, by 50 days, degenerative changes are evident in the saccular macula, which progresses to total loss of the saccular neuroepithelium in older animals. The new allele of av reported here will be designated av(TgN2742Rpw). (C) 2000 Elsevier Science B.V. All rights reserved.

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Documento generato il 25/11/20 alle ore 06:29:10