The DOI matches its Crossref record; 1 of the 1 references it cites were found in a registry or an index.
Germ cell defects and hematopoietic hypersensitivity to gamma-interferon in mice with a targeted disruption of the Fanconi anemia C gene
Abstract
Fanconi anemia (FA) is an autosomal recessive chromosome instability syndrome characterized by progressive bone marrow (BM) failure, skeletal defects, and increased susceptibility to malignancy. FA cells are hypersensitive to DNA cross-linking agents, oxygen and have cell cycle abnormalities. To develop an animal model of the disease we generated mice homozygous for a targeted deletion of exon 9 of the murine FA complementation group C gene (fac). Mutant mice had normal neonatal viability and gross morphology, but their cells had the expected chromosome breakage and DNA cross-linker sensitivity. Surprisingly, male and female mutant mice had reduced numbers of germ cells and females had markedly impaired fertility. No anemia was detectable in the peripheral blood during the first year of life, but the colony forming capacity of marrow progenitor cells was abnormal in vitro in mutant mice. Progenitor cells from fac knock-out mice were hypersensitive to interferon gamma. This previously unrecognized phenotype may form the basis for BM failure in human FA.
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“In Fancc−/− mice, TNF-α and INF-γ induce apoptosis in hematopoietic progenitor cells, even at low cytokine levels[16–18].”
From Finding the needle in the hay stack: Hematopoietic stem cells in Fanconi anemia (2009) · cites this paper as [17]
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Zoumbos, N. C., Gascon, P., Djeu, J. Y., & Young, N. S. (1985) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.82.1.188 · Cited by 330
Research works printed but not linked: 2
- Bradley A: Production and analysis of chimaeric mice, in Robertson EJ (eds): Teratocarcinomas and Embryonic Stem Cells. A Practical Approach. Oxford, UK, IRL, 1987, p 113
- Silvers WK: The Coat Colors of Mice: A Model for Mamma- lian Gene Action and Interaction. New York, NY Springer-Verlag. 1979
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Interaction of the Fanconi Anemia Proteins and BRCA1 in a Common Pathway
Garcia-Higuera, I., Taniguchi, T., Ganesan, S., Meyn, M. S., Timmers, C., Hejna, J., Grompe, M., & D'Andrea, A. D. (2001) · Molecular Cell · doi:10.1016/s1097-2765(01)00173-3 · Cited by 1,198
Self-citationThe Fanconi anaemia/BRCA pathway
D'Andrea, A. D., & Grompe, M. (2003) · Nature Reviews Cancer · doi:10.1038/nrc970 · Cited by 822
Self-citationExit from dormancy provokes DNA-damage-induced attrition in haematopoietic stem cells
Walter, D., Lier, A., Geiselhart, A., Thalheimer, F. B., Huntscha, S., Sobotta, M. C., Moehrle, B., Brocks, D., Bayindir, I., Kaschutnig, P., et al. (2015) · Nature · doi:10.1038/nature14131 · Cited by 619
Recombinational DNA repair and human disease
Thompson, L. H., & Schild, D. (2002) · Mutation Research - Fundamental and Molecular Mechanisms of Mutagenesis · doi:10.1016/s0027-5107(02)00224-5 · Cited by 417
Hematopoietic stem cell defects in mice with deficiency of Fancd2 or Usp1
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Oxidative Stress, Bone Marrow Failure, and Genome Instability in Hematopoietic Stem Cells
Richardson, C., Yan, S., & Vestal, C. (2015) · International Journal of Molecular Sciences · doi:10.3390/ijms16022366 · Cited by 81
In vivo selection of wild‐type hematopoietic stem cells in a murine model of Fanconi anemia
Battaile, K. P., Bateman, R. L., Mortimer, D., Mulcahy, J., Rathbun, R. K., Bagby, G., Fleming, W. H., & Grompe, M. (1999) · Blood · doi:10.1182/blood.v94.6.2151 · Cited by 54
Self-citationMyelodysplastic syndrome: An inability to appropriately respond to damaged DNA?
Zhou, T., Hasty, P., Walter, C. A., Bishop, A. J. R., Scott, L. M., & Rebel, V. I. (2013) · Experimental Hematology · doi:10.1016/j.exphem.2013.04.008 · Cited by 39
Finding the needle in the hay stack: Hematopoietic stem cells in Fanconi anemia
Müller, L. U. W., & Williams, D. A. (2009) · Mutation Research - Fundamental and Molecular Mechanisms of Mutagenesis · doi:10.1016/j.mrfmmm.2009.03.010 · Cited by 31
Metadata
- Authors
- Whitney, M., Royle, G., Low, M., Kelly, M., Axthelm, M., Reifsteck, C., Olson, S., Braun, R., Heinrich, M., Rathbun, R., Bagby, G., & Grompe, M.
- Affiliations
- Department of Molecular and Medical Genetics, Vollum Institute, Oregon Regional Primate Center, Portland, USA
- Venue
- Blood
- Publisher
- American Society of Hematology
- Published
- 1996-07-01
- Volume
- 88
- Issue
- 1
- Pages
- 49-58
- ISSN
- 0006-4971
- 1528-0020
- Cited by
- 229
- References
- 1
- Language
- en
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