Germ cell aneuploidy in zebrafish with mutations in the mitotic checkpoint gene mps1
- Kenneth D. Poss1,2,4,7,10,
- Alex Nechiporuk1,2,4,8,
- Keith F. Stringer1,2,4,5,9,
- Charles Lee3,6, and
- Mark T. Keating1,2,4
- 1Howard Hughes Medical Institute, 2Department of Cell Biology and 3Department of Pathology, Harvard Medical School, Boston, Massachusetts 02115, USA; 4Department of Cardiology and 5Department of Pathology, Children's Hospital, Boston, Massachusetts 02115, USA; 6Department of Pathology, Brigham and Women's Hospital, Boston, Massachusetts 02115, USA; 7Department of Cell Biology, Duke University Medical Center, Durham, North Carolina 27710, USA
Abstract
Aneuploidy, resulting from chromosome missegregation during meiosis, is a major cause of human infertility and birth defects. However, its molecular basis remains incompletely understood. Here we have identified a spectrum of chromosome anomalies in embryos of zebrafish homozygous for a hypomorphic mutation in Mps1, a kinase required for the mitotic checkpoint. These aneuploidies are caused by meiotic error and result in severe developmental defects. Our results reveal Mps1 as a critical regulator of chromosome number in zebrafish, and demonstrate how slight genetic perturbation of a mitotic checkpoint factor can dramatically reduce the fidelity of chromosome segregation during vertebrate meiosis.
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Footnotes
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Article and publication are at http://www.genesdev.org/cgi/doi/10.1101/gad.1182604.
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↵10 Corresponding author. E-MAIL k.poss{at}cellbio.duke.edu; FAX (919) 684-5481.
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↵8 Present address: Department of Biological Structure, University of Washington, Seattle, WA 98195, USA
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↵9 Present address: Department of Pathology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229, USA.
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- Accepted May 11, 2004.
- Received December 30, 2003.
- Cold Spring Harbor Laboratory Press
