期刊
FASEB JOURNAL
卷 23, 期 7, 页码 2215-2225出版社
WILEY
DOI: 10.1096/fj.08-123737
关键词
ER stress; RNAi; deletion mutant; glycosylation; model organism
资金
- Japan Science and Technology Corp
- MEXT, Japan
Although the solute carrier 35B1 (SLC35B1) is evolutionarily conserved, its functions in metazoans remain unknown. To elucidate its function, we examined developmental roles of an SLC35B1 family gene (HUT-1: homolog of UDP-Gal transporter) in Caenorhabditis elegans. We isolated a deletion mutant of the gene and characterized phenotypes of the mutant and hut-1 RNAi-treated worms. GFP-HUT-1 reporter analysis was performed to examine gene expression patterns. We also tested whether several nucleotide sugar transporters can compensate for hut-1 deficiency. The hut-1 deletion mutant and RNAi worms showed larval growth defect and lethality with disrupted intestinal morphology. Inactivation of hut-1 induced chronic endoplasmic reticulum ( ER) stress, and hut-1 showed genetic interactions with the atf-6, pek-1, and ire-1 genes involved in unfolded protein response signaling. ER ultrastructure and ER marker distribution in hut-1-deficient animals showed that HUT-1 is required for maintenance of ER structure. Reporter analysis revealed that HUT- 1 is an ER protein ubiquitously expressed in tissues, including the intestine. Lethality and the ER stress phenotype of the mutant were rescued with the human hut-1 ortholog UGTrel1. These results indicate important roles for hut-1 in development and maintenance of ER homeostasis in C. elegans.-Dejima, K., Murata, D., Mizuguchi, S., Nomura, K. H., Gengyo-Ando, K., Mitani, S., Kamiyama, S., Nishihara, S., Nomura, K. The ortholog of human solute carrier family 35 member B1 (UDP-galactose transporter- related protein 1) is involved in maintenance of ER homeostasis and essential for larval development in Caenorhabditis elegans. FASEB J. 23, 2215-2225 (2009)
作者
我是这篇论文的作者
点击您的名字以认领此论文并将其添加到您的个人资料中。
推荐
暂无数据