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Targeted disruption of NDST‐1 gene leads to pulmonary hypoplasia and neonatal respiratory distress in mice
Abstract
In order to address the biological function of GlcNAc N‐deacetylase/N‐sulfotransferase‐1 (NDST‐1), we disrupted the NDST‐1 gene by homologous recombination in mouse embryonic stem cells. The NDST‐1 null mice developed respiratory distress and atelectasis that subsequently caused neonatal death. Morphological examination revealed type II pneumocyte immaturity, which was characterized by an increased glycogen content and a reduced number of lamellar bodies and microvilli. Biochemical analysis further indicated that both total phospholipids and disaturated phosphatidylcholine were reduced in the mutant lung. Our data revealed that NDST‐1 was essential for the maturation of type II pneumocytes and its inactivation led to a neonatal respiratory distress syndrome.
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Cites
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Li, E., Bestor, T. H., & Jaenisch, R. (1992) · Cell · doi:10.1016/0092-8674(92)90611-f · Cited by 3,984
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Bishop, J. R., Schuksz, M., & Esko, J. D. (2007) · Nature · doi:10.1038/nature05817 · Cited by 1,631
Sarrazin, S., Lamanna, W. C., & Esko, J. D. (2011) · Cold Spring Harbor Perspectives in Biology · doi:10.1101/cshperspect.a004952 · Cited by 1,516
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Esko, J. D., & Selleck, S. B. (2002) · Annual Review of Biochemistry · doi:10.1146/annurev.biochem.71.110601.135458 · Cited by 1,495
A Genetic Approach to Mammalian Glycan Function
Lowe, J. B., & Marth, J. D. (2003) · Annual Review of Biochemistry · doi:10.1146/annurev.biochem.72.121801.161809 · Cited by 594
Sulfonation and molecular action
Strott, C. A. (2002) · Endocrine Reviews · doi:10.1210/er.2001-0040 · Cited by 487
Metadata
- Authors
- Fan, G., Xiao, L., Cheng, L., Wang, X., Sun, B., & Hu, G.
- Venue
- FEBS Letters
- Publisher
- Wiley
- Published
- 2000-02-02
- Volume
- 467
- Issue
- 1
- Pages
- 7-11
- ISSN
- 0014-5793 (ISSN-L)
- 1873-3468
- OpenAlex
- W1976962628
- Cited by
- 165
- References
- 1
- FWCI
- 10.02
- Type
- journal article
- Language
- en
- Open access
- closed
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- The paper
- Fan, G., Xiao, L., Cheng, L., Wang, X., Sun, B., & Hu, G. (2000). Targeted disruption of NDST‐1 gene leads to pulmonary hypoplasia and neonatal respiratory distress in mice. FEBS Letters. https://doi.org/10.1016/s0014-5793(00)01111-x
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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.
