| RRC ID |
6447
|
| Author |
Dejima K, Murata D, Mizuguchi S, Nomura KH, Gengyo-Ando K, Mitani S, Kamiyama S, Nishihara S, Nomura K.
|
| 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 |
FASEB J
|
| Abstract |
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.
|
| Volume |
23(7)
|
| Pages |
2215-25
|
| Published |
2009-7-1
|
| DOI |
10.1096/fj.08-123737
|
| PII |
fj.08-123737
|
| PMID |
19270184
|
| PMC |
PMC2704591
|
| MeSH |
Animals
Caenorhabditis elegans / physiology*
Endoplasmic Reticulum / physiology*
Gene Expression Regulation
Genes, Reporter
Homeostasis*
Larva / growth & development*
Monosaccharide Transport Proteins / physiology*
Nucleotide Transport Proteins
Phenotype
RNA, Small Interfering / pharmacology
Sequence Deletion
|
| IF |
4.966
|
| Times Cited |
12
|
|
WOS Category
|
BIOLOGY
BIOCHEMISTRY & MOLECULAR BIOLOGY
CELL BIOLOGY
|
| Resource |
| C.elegans |
tm1435 |
