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Publication : LUZP deficiency affects neural tube closure during brain development.

First Author  Hsu CY Year  2008
Journal  Biochem Biophys Res Commun Volume  376
Issue  3 Pages  466-71
PubMed ID  18801334 Mgi Jnum  J:141306
Mgi Id  MGI:3818102 Doi  10.1016/j.bbrc.2008.08.170
Citation  Hsu CY, et al. (2008) LUZP deficiency affects neural tube closure during brain development. Biochem Biophys Res Commun 376(3):466-71
abstractText  LUZP is a leucine zipper-containing protein predominantly expressed in the brain. The functional significance of LUZP remains unknown. To explore the role of LUZP in brain development, a knockout mouse strain with a lacZ knock-in (Luzp-KO/lacZ-KI) has been established. LacZ reporter expression driven by the endogenous Luzp promoter was detected in the neuroepithelium and the cardiac tissue. Luzp(-/-) mice exhibited perinatal death, presumably due to the accompanied complex cardiovascular defects. Luzp(-/-) embryos displayed a cranial neural tube closure defect (NTD), with exposed brain tissues. Ectopic expression of Sonic-hedgehog, which is a protein known to be involved in neural tube closure, and elevated apoptosis were observed in the dorsal lateral neuroepithelium of the NTD Luzp(-/-) hindbrain. These findings assign a novel function of LUZP in the embryonic development of brain.
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