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Essential Role of Fkbp6 in Male Fertility and Homologous Chromosome Pairing in Meiosis
Open accessAbstract
Meiosis is a critical stage of gametogenesis in which alignment and synapsis of chromosomal pairs occur, allowing for the recombination of maternal and paternal genomes. Here we show that FK506 binding protein (Fkbp6) localizes to meiotic chromosome cores and regions of homologous chromosome synapsis. Targeted inactivation of Fkbp6 in mice results in aspermic males and the absence of normal pachytene spermatocytes. Moreover, we identified the deletion of Fkbp6 exon 8 as the causative mutation in spontaneously male sterile as / as mutant rats. Loss of Fkbp6 results in abnormal pairing and misalignments between homologous chromosomes, nonhomologous partner switches, and autosynapsis of X chromosome cores in meiotic spermatocytes. Fertility and meiosis are normal in Fkbp6 mutant females. Thus, Fkbp6 is a component of the synaptonemal complex essential for sex-specific fertility and for the fidelity of homologous chromosome pairing in meiosis.
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X-linked Gene for Testicular Feminization in the Mouse
LYON, M. F., & HAWKES, S. G. (1970) · Nature · doi:10.1038/2271217a0 · Cited by 555
Cited by
Absence of Mouse REC8 Cohesin Promotes Synapsis of Sister Chromatids in Meiosis
Xu, H., Beasley, M. D., Warren, W. D., van der Horst, G. T. J., & McKay, M. J. (2005) · Developmental Cell · doi:10.1016/j.devcel.2005.03.018 · Cited by 351
Strogantsev, R., Krueger, F., Yamazawa, K., Shi, H., Gould, P., Goldman-Roberts, M., McEwen, K., Sun, B., Pedersen, R., & Ferguson-Smith, A. C. (2015) · Genome Biology · doi:10.1186/s13059-015-0672-7 · Cited by 152
Proulx, É., Young, E. J., Osborne, L. R., & Lambe, E. K. (2010) · Journal of Neurodevelopmental Disorders · doi:10.1007/s11689-010-9044-5 · Cited by 35
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Metadata
- Authors
- Crackower, M. A., Kolas, N. K., Noguchi, J., Sarao, R., Kikuchi, K., Kaneko, H., Kobayashi, E., Kawai, Y., Kozieradzki, I., Landers, R., Mo, R., Hui, C.-C., Nieves, E., Cohen, P. E., Osborne, L. R., Wada, T., Kunieda, T., Moens, P. B., & Penninger, J. M.
- Venue
- Science
- Published
- 2003-05-23
- Volume
- 300
- Issue
- 5623
- Pages
- 1291-1295
- ISSN
- 0036-8075 (ISSN-L)
- 1095-9203
- OpenAlex
- W2111893619
- Cited by
- 216
- References
- 1
- FWCI
- 14.27
- Type
- journal article
- Language
- en
- Open access
- green
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- Crackower, M. A., Kolas, N. K., Noguchi, J., Sarao, R., Kikuchi, K., Kaneko, H., Kobayashi, E., Kawai, Y., Kozieradzki, I., Landers, R., Mo, R., Hui, C.-C., Nieves, E., Cohen, P. E., Osborne, L. R., Wada, T., Kunieda, T., Moens, P. B., & Penninger, J. M. (2003). Essential Role of Fkbp6 in Male Fertility and Homologous Chromosome Pairing in Meiosis. Science. https://doi.org/10.1126/science.1083022
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Contains information from the Semantic Scholar Academic Graph Datasets, made available under the ODC Attribution License (ODC-BY 1.0).
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