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Correction of Deafness in shaker-2 Mice by an Unconventional Myosin in a BAC Transgene
Abstract
The shaker-2 mouse mutation, the homolog of human DFNB3 , causes deafness and circling behavior. A bacterial artificial chromosome (BAC) transgene from the shaker-2 critical region corrected the vestibular defects, deafness, and inner ear morphology of shaker-2 mice. An unconventional myosin gene, Myo15 , was discovered by DNA sequencing of this BAC. Shaker-2 mice were found to have an amino acid substitution at a highly conserved position within the motor domain of this myosin. Auditory hair cells of shaker-2 mice have very short stereocilia and a long actin-containing protrusion extending from their basal end. This histopathology suggests that Myo15 is necessary for actin organization in the hair cells of the cochlea.
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Citations
Cited by
Role of Myosin VI in the Differentiation of Cochlear Hair Cells
Self, T., Sobe, T., Copeland, N. G., Jenkins, N. A., Avraham, K. B., & Steel, K. P. (1999) · Developmental Biology · doi:10.1006/dbio.1999.9424 · Cited by 263
Sprunger, L. (1999) · Human Molecular Genetics · doi:10.1093/hmg/8.3.471 · Cited by 72
Structural basis for tunable control of actin dynamics by myosin-15 in mechanosensory stereocilia
Gong, R., Jiang, F., Moreland, Z. G., Reynolds, M. J., de los Reyes, S. E., Gurel, P., Shams, A., Heidings, J. B., Bowl, M. R., Bird, J. E., et al. (2022) · Science Advances · doi:10.1126/sciadv.abl4733 · Cited by 56
Metadata
- Authors
- Probst, F. J., Fridell, R. A., Raphael, Y., Saunders, T. L., Wang, A., Liang, Y., Morell, R. J., Touchman, J. W., Lyons, R. H., Noben-Trauth, K., Friedman, T. B., & Camper, S. A.
- Venue
- Science
- Published
- 1998-05-29
- Volume
- 280
- Issue
- 5368
- Pages
- 1444-1447
- ISSN
- 0036-8075 (ISSN-L)
- 1095-9203
- OpenAlex
- W2130986890
- Cited by
- 414
- References
- 0
- Type
- journal article
- Language
- en
- Open access
- closed
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- Probst, F. J., Fridell, R. A., Raphael, Y., Saunders, T. L., Wang, A., Liang, Y., Morell, R. J., Touchman, J. W., Lyons, R. H., Noben-Trauth, K., Friedman, T. B., & Camper, S. A. (1998). Correction of Deafness in shaker-2 Mice by an Unconventional Myosin in a BAC Transgene. Science. https://doi.org/10.1126/science.280.5368.1444
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Contains information from the Semantic Scholar Academic Graph Datasets, made available under the ODC Attribution License (ODC-BY 1.0).
Semantic Scholar’s data is described in: Kinney, R., et al. (2023). The Semantic Scholar Open Data Platform. arXiv:2301.10140.
