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

Katsufumi Dejima, Daisuke Murata, Souhei Mizuguchi, Kazuko H. Nomura, Keiko Gengyo-Ando, Shohei Mitani, Shin Kamiyama, Shoko Nishihara, Kazuya Nomura

Research output: Contribution to journalArticle

12 Citations (Scopus)

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 RNAitreated 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.

Original languageEnglish
Pages (from-to)2215-2225
Number of pages11
JournalFASEB Journal
Volume23
Issue number7
DOIs
Publication statusPublished - Jul 1 2009

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Caenorhabditis elegans
Endoplasmic Reticulum
Homeostasis
Genes
Maintenance
Endoplasmic Reticulum Stress
Proteins
Gene expression
Sugars
Animals
Phenotype
Unfolded Protein Response
Nucleotides
Tissue
Gene Deletion
Defects
RNA Interference
Intestines
UDP-galactose translocator
Gene Expression

All Science Journal Classification (ASJC) codes

  • Biotechnology
  • Biochemistry
  • Molecular Biology
  • Genetics

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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. / Dejima, Katsufumi; Murata, Daisuke; Mizuguchi, Souhei; Nomura, Kazuko H.; Gengyo-Ando, Keiko; Mitani, Shohei; Kamiyama, Shin; Nishihara, Shoko; Nomura, Kazuya.

In: FASEB Journal, Vol. 23, No. 7, 01.07.2009, p. 2215-2225.

Research output: Contribution to journalArticle

Dejima, Katsufumi ; Murata, Daisuke ; Mizuguchi, Souhei ; Nomura, Kazuko H. ; Gengyo-Ando, Keiko ; Mitani, Shohei ; Kamiyama, Shin ; Nishihara, Shoko ; Nomura, Kazuya. / 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. In: FASEB Journal. 2009 ; Vol. 23, No. 7. pp. 2215-2225.
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