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Dejima Katsufumi
Department School of Medicine, School of Medicine Position Assistant Professor |
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| Article types | Original article |
| Language | English |
| Peer review | Peer reviewed |
| Title | 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. |
| Journal | Formal name：FASEB journal : official publication of the Federation of American Societies for Experimental Biology Abbreviation：FASEB J ISSN code：1530-6860(Electronic)0892-6638(Linking) |
| Volume, Issue, Page | 23(7),pp.2215-25 |
| Author and coauthor | Dejima Katsufumi, Murata Daisuke, Mizuguchi Souhei, Nomura Kazuko H, Gengyo-Ando Keiko, Mitani Shohei, Kamiyama Shin, Nishihara Shoko, Nomura Kazuya |
| Publication date | 2009/07 |
| Summary | 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. |
| DOI | 10.1096/fj.08-123737 |
| Document No. | 19270184 |