| First Author | Yagishita N | Year | 2005 |
| Journal | J Biol Chem | Volume | 280 |
| Issue | 9 | Pages | 7909-16 |
| PubMed ID | 15611074 | Mgi Jnum | J:97254 |
| Mgi Id | MGI:3575050 | Doi | 10.1074/jbc.M410863200 |
| Citation | Yagishita N, et al. (2005) Essential role of synoviolin in embryogenesis. J Biol Chem 280(9):7909-16 |
| abstractText | We recently reported the importance of Synoviolin in quality control of proteins through the endoplasmic reticulum (ER)-associated degradation (ERAD) system and its involvement in the pathogenesis of arthropathy through its anti-apoptotic effect. For further understanding of the role of Synoviolin in vivo, we generated in this study synoviolin-deficient (syno(-/-)) mice by genetargeted disruption. Strikingly, all fetuses lacking syno died in utero around embryonic day 13.5, although Hrd1p, a yeast orthologue of Synoviolin, is non-essential for survival. Histologically, hypocellularity and aberrant apoptosis were noted in the syno(-/-) fetal liver. Moreover, definitive erythropoiesis was affected in non-cell autonomous manner in syno(-/-) embryos, causing death in utero. Cultured embryonic fibroblasts derived from syno(-/-) mice were more susceptible to endoplasmic reticulum stress-induced apoptosis than those from syno(+/+) mice, but the susceptibility was rescued by overexpression of synoviolin. Our findings emphasized the indispensable role of the Synoviolin in embryogenesis. |
