Literature DB >> 12760743

Cloning of an emopamil-binding protein (EBP)-like protein that lacks sterol delta8-delta7 isomerase activity.

Fabian F Moebius1, Barbara U Fitzky, Georg Wietzorrek, Alexander Haidekker, Andrea Eder, Hartmut Glossmann.   

Abstract

EBP (emopamil-binding protein) is a high-affinity binding protein for [3H]emopamil and belongs to the family of so-called sigma receptors. Mutations that disrupt EBP's 3beta-hydroxysteroid sterol delta8-delta7 isomerase activity (EC 5.3.3.5) impair cholesterol biosynthesis and cause X-chromosomal dominant chondrodysplasia punctata. We identified a human cDNA for a novel EBPL (EBP-like protein) with a calculated mass of 23.2 kDa. Amino acid sequence alignments and phylogenetic analysis revealed that EBPL is distantly related to EBP (31% identity and 52% similarity) and found in animals but not in plants. EBPL is encoded by four exons on human chromosome 13q14.2 covering 30.7 kb, and a partially processed EBPL pseudogene was found on 16q21. The EBPL mRNA was expressed ubiquitously and most abundant in liver, lung and kidney. Upon heterologous expression in yeast EBPL had no detectable 3beta-hydroxysteroid sterol delta8-delta7 isomerase and sigma-ligand-binding activity. Nine out of ten amino acid residues essential for catalytic activity of EBP were conserved in EBPL. Replacement of the only differing residue (EBP-Y111W) reduced catalytic activity of EBP. Transfer of the divergent residue from EBP to EBPL (EBPL-W91Y) and chimaerization of EBP and EBPL at various positions failed to restore catalytic activity of EBPL. Chemical cross-linking induced homodimerization of EBPL and EBP. Whereas mevinolin increased the mRNA for EBP and DHCR7 (delta7-sterol reductase) in HepG2 cells, it had no effect on mRNAs for EBPL and sigma1 receptor, indicating that EBP and EBPL expression are not co-ordinated. We propose that EBPL has a yet-to-be-discovered function other than cholesterol biosynthesis.

Entities:  

Mesh:

Substances:

Year:  2003        PMID: 12760743      PMCID: PMC1223579          DOI: 10.1042/BJ20030465

Source DB:  PubMed          Journal:  Biochem J        ISSN: 0264-6021            Impact factor:   3.857


  30 in total

1.  Isolation and characterization of an Arabidopsis thaliana C-8,7 sterol isomerase: functional and structural similarities to mammalian C-8,7 sterol isomerase/emopamil-binding protein.

Authors:  R J Grebenok; T E Ohnmeiss; A Yamamoto; E D Huntley; D W Galbraith; D Della Penna
Journal:  Plant Mol Biol       Date:  1998-11       Impact factor: 4.076

2.  Histidine77, glutamic acid81, glutamic acid123, threonine126, asparagine194, and tryptophan197 of the human emopamil binding protein are required for in vivo sterol delta 8-delta 7 isomerization.

Authors:  F F Moebius; K E Soellner; B Fiechtner; C W Huck; G Bonn; H Glossmann
Journal:  Biochemistry       Date:  1999-01-19       Impact factor: 3.162

3.  Statistical features of human exons and their flanking regions.

Authors:  M Q Zhang
Journal:  Hum Mol Genet       Date:  1998-05       Impact factor: 6.150

Review 4.  The mysteries of sigma receptors: new family members reveal a role in cholesterol synthesis.

Authors:  F F Moebius; J Striessnig; H Glossmann
Journal:  Trends Pharmacol Sci       Date:  1997-03       Impact factor: 14.819

5.  The gene mutated in bare patches and striated mice encodes a novel 3beta-hydroxysteroid dehydrogenase.

Authors:  X Y Liu; A W Dangel; R I Kelley; W Zhao; P Denny; M Botcherby; B Cattanach; J Peters; P R Hunsicker; A M Mallon; M A Strivens; R Bate; W Miller; M Rhodes; S D Brown; G E Herman
Journal:  Nat Genet       Date:  1999-06       Impact factor: 38.330

6.  Pharmacological analysis of sterol delta8-delta7 isomerase proteins with [3H]ifenprodil.

Authors:  F F Moebius; R J Reiter; K Bermoser; H Glossmann; S Y Cho; Y K Paik
Journal:  Mol Pharmacol       Date:  1998-09       Impact factor: 4.436

7.  Purification, molecular cloning, and expression of the mammalian sigma1-binding site.

Authors:  M Hanner; F F Moebius; A Flandorfer; H G Knaus; J Striessnig; E Kempner; H Glossmann
Journal:  Proc Natl Acad Sci U S A       Date:  1996-07-23       Impact factor: 11.205

8.  Mutations in the Delta7-sterol reductase gene in patients with the Smith-Lemli-Opitz syndrome.

Authors:  B U Fitzky; M Witsch-Baumgartner; M Erdel; J N Lee; Y K Paik; H Glossmann; G Utermann; F F Moebius
Journal:  Proc Natl Acad Sci U S A       Date:  1998-07-07       Impact factor: 11.205

9.  Diagnosis of Smith-Lemli-Opitz syndrome by gas chromatography/mass spectrometry of 7-dehydrocholesterol in plasma, amniotic fluid and cultured skin fibroblasts.

Authors:  R I Kelley
Journal:  Clin Chim Acta       Date:  1995-04-30       Impact factor: 3.786

10.  Human lamin B receptor exhibits sterol C14-reductase activity in Saccharomyces cerevisiae.

Authors:  S Silve; P H Dupuy; P Ferrara; G Loison
Journal:  Biochim Biophys Acta       Date:  1998-06-15
View more
  9 in total

1.  The Effect of Small Molecules on Sterol Homeostasis: Measuring 7-Dehydrocholesterol in Dhcr7-Deficient Neuro2a Cells and Human Fibroblasts.

Authors:  Zeljka Korade; Hye-Young H Kim; Keri A Tallman; Wei Liu; Katalin Koczok; Istvan Balogh; Libin Xu; Karoly Mirnics; Ned A Porter
Journal:  J Med Chem       Date:  2016-01-29       Impact factor: 7.446

2.  A Botrytis cinerea emopamil binding domain protein, required for full virulence, belongs to a eukaryotic superfamily which has expanded in euascomycetes.

Authors:  A Gioti; J M Pradier; E Fournier; P Le Pêcheur; C Giraud; D Debieu; J Bach; P Leroux; C Levis
Journal:  Eukaryot Cell       Date:  2007-12-21

3.  Expression of targeted ribozyme against telomerase RNA causes altered expression of several other genes in tumor cells.

Authors:  Suresh Kumar Ramakrishnan; Akhil Varshney; Amod Sharma; Bhudev C Das; Pramod K Yadava
Journal:  Tumour Biol       Date:  2014-03-25

4.  Identification of candidate epigenetic biomarkers for ovarian cancer detection.

Authors:  Yi-Wen Huang; Rachel A Jansen; Enrica Fabbri; Dustin Potter; Sandya Liyanarachchi; Michael W Y Chan; Joseph C Liu; Anne P G Crijns; Robert Brown; Kenneth P Nephew; Ate G J van der Zee; David E Cohn; Pearlly S Yan; Tim H-M Huang; Huey-Jen L Lin
Journal:  Oncol Rep       Date:  2009-10       Impact factor: 3.906

5.  Genotype-phenotype correlations of Berardinelli-Seip congenital lipodystrophy and novel candidate genes prediction.

Authors:  Meng Ren; Jingru Shi; Jinmeng Jia; Yongli Guo; Xin Ni; Tieliu Shi
Journal:  Orphanet J Rare Dis       Date:  2020-04-29       Impact factor: 4.123

6.  Transcriptome-wide association study identifies susceptibility genes for rheumatoid arthritis.

Authors:  Cuiyan Wu; Sijian Tan; Li Liu; Shiqiang Cheng; Peilin Li; Wenyu Li; Huan Liu; Feng'e Zhang; Sen Wang; Yujie Ning; Yan Wen; Feng Zhang
Journal:  Arthritis Res Ther       Date:  2021-01-22       Impact factor: 5.156

7.  Investigation of the Molecular Mechanisms of Antioxidant Damage and Immune Response Downregulation in Liver of Coilia nasus Under Starvation Stress.

Authors:  Meiyao Wang; Gangchun Xu; Yongkai Tang; Shengyan Su; Yinping Wang; Zhixiang Zhu
Journal:  Front Endocrinol (Lausanne)       Date:  2021-02-26       Impact factor: 5.555

8.  Amiodarone Alters Cholesterol Biosynthesis through Tissue-Dependent Inhibition of Emopamil Binding Protein and Dehydrocholesterol Reductase 24.

Authors:  Luke B Allen; Thiago C Genaro-Mattos; Allison Anderson; Ned A Porter; Károly Mirnics; Zeljka Korade
Journal:  ACS Chem Neurosci       Date:  2020-04-29       Impact factor: 4.418

9.  Sterol metabolism in the filasterean Capsaspora owczarzaki has features that resemble both fungi and animals.

Authors:  Sebastián R Najle; María Celeste Molina; Iñaki Ruiz-Trillo; Antonio D Uttaro
Journal:  Open Biol       Date:  2016-07       Impact factor: 6.411

  9 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.