Literature DB >> 1430210

Animal cell mutants represent two complementation groups of peroxisome-defective Zellweger syndrome.

N Shimozawa1, T Tsukamoto, Y Suzuki, T Orii, Y Fujiki.   

Abstract

Generalized peroxisome-deficient disorders including cerebro-hepato-renal Zellweger syndrome, neonatal adrenoleukodystrophy, and infantile Refsum disease are autosomal recessive diseases, where catalase-containing particles (peroxisomes) are morphologically absent. We previously isolated two Chinese hamster ovary (CHO) cell mutants (Z24 and Z65) that resemble the fibroblasts from patients with such diseases, in their defective peroxisome assembly (Tsukamoto, T., S. Yokota, and Y. Fujiki. 1990. J. Cell Biol. 110:651-660). Here we report isolation by the P9OH/UV method of a peroxisome-deficient CHO mutant, ZP92, of the third complementation group distinct from those of Z24 and Z65. Peroxisomal membrane ghosts were noted by immunochemical staining in all of the CHO mutants. Complementation analysis by cell fusion of the CHO mutants with cultured fibroblasts from patients with generalized peroxisomal disorders revealed that two CHO mutants (Z24 and ZP92) represent the human complementation groups, E (the same as group 1 in the U.S.) and C (the same as group 4), respectively. These CHO cell mutants are an apparently relevant animal cell model for studies on the molecular bases and primary defects of human peroxisome-deficient diseases.

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Year:  1992        PMID: 1430210      PMCID: PMC443247          DOI: 10.1172/JCI116063

Source DB:  PubMed          Journal:  J Clin Invest        ISSN: 0021-9738            Impact factor:   14.808


  21 in total

1.  Complementation study of peroxisome-deficient disorders by immunofluorescence staining and characterization of fused cells.

Authors:  S Yajima; Y Suzuki; N Shimozawa; S Yamaguchi; T Orii; Y Fujiki; T Osumi; T Hashimoto; H W Moser
Journal:  Hum Genet       Date:  1992-03       Impact factor: 4.132

2.  Complete nucleotide sequence of cDNA and predicted amino acid sequence of rat acyl-CoA oxidase.

Authors:  S Miyazawa; H Hayashi; M Hijikata; N Ishii; S Furuta; H Kagamiyama; T Osumi; T Hashimoto
Journal:  J Biol Chem       Date:  1987-06-15       Impact factor: 5.157

3.  Genetic and phenotypic heterogeneity in disorders of peroxisome biogenesis--a complementation study involving cell lines from 19 patients.

Authors:  A A Roscher; S Hoefler; G Hoefler; E Paschke; F Paltauf; A Moser; H Moser
Journal:  Pediatr Res       Date:  1989-07       Impact factor: 3.756

4.  A presumed DNA helicase encoded by ERCC-3 is involved in the human repair disorders xeroderma pigmentosum and Cockayne's syndrome.

Authors:  G Weeda; R C van Ham; W Vermeulen; D Bootsma; A J van der Eb; J H Hoeijmakers
Journal:  Cell       Date:  1990-08-24       Impact factor: 41.582

5.  A rapid selection for animal cell mutants with defective peroxisomes.

Authors:  O H Morand; L A Allen; R A Zoeller; C R Raetz
Journal:  Biochim Biophys Acta       Date:  1990-05-16

6.  A human gene responsible for Zellweger syndrome that affects peroxisome assembly.

Authors:  N Shimozawa; T Tsukamoto; Y Suzuki; T Orii; Y Shirayoshi; T Mori; Y Fujiki
Journal:  Science       Date:  1992-02-28       Impact factor: 47.728

7.  Restoration by a 35K membrane protein of peroxisome assembly in a peroxisome-deficient mammalian cell mutant.

Authors:  T Tsukamoto; S Miura; Y Fujiki
Journal:  Nature       Date:  1991-03-07       Impact factor: 49.962

8.  Synthesis of 3-ketoacyl-CoA thiolase of rat liver peroxisomes on free polyribosomes as a larger precursor. Induction of thiolase mRNA activity by clofibrate.

Authors:  Y Fujiki; R A Rachubinski; R M Mortensen; P B Lazarow
Journal:  Biochem J       Date:  1985-03-15       Impact factor: 3.857

9.  Isolation and characterization of Chinese hamster ovary cell mutants defective in assembly of peroxisomes.

Authors:  T Tsukamoto; S Yokota; Y Fujiki
Journal:  J Cell Biol       Date:  1990-03       Impact factor: 10.539

10.  PAS3, a Saccharomyces cerevisiae gene encoding a peroxisomal integral membrane protein essential for peroxisome biogenesis.

Authors:  J Höhfeld; M Veenhuis; W H Kunau
Journal:  J Cell Biol       Date:  1991-09       Impact factor: 10.539

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  17 in total

1.  Phenotype-genotype relationships in peroxisome biogenesis disorders of PEX1-defective complementation group 1 are defined by Pex1p-Pex6p interaction.

Authors:  S Tamura; N Matsumoto; A Imamura; N Shimozawa; Y Suzuki; N Kondo; Y Fujiki
Journal:  Biochem J       Date:  2001-07-15       Impact factor: 3.857

Review 2.  Peroxisome Biogenesis Disorders.

Authors:  Masanori Honsho; Kanji Okumoto; Shigehiko Tamura; Yukio Fujiki
Journal:  Adv Exp Med Biol       Date:  2020       Impact factor: 2.622

3.  Deficiency of a Retinal Dystrophy Protein, Acyl-CoA Binding Domain-containing 5 (ACBD5), Impairs Peroxisomal β-Oxidation of Very-long-chain Fatty Acids.

Authors:  Yuichi Yagita; Kyoko Shinohara; Yuichi Abe; Keiko Nakagawa; Mohammed Al-Owain; Fowzan S Alkuraya; Yukio Fujiki
Journal:  J Biol Chem       Date:  2016-11-29       Impact factor: 5.157

4.  Human PEX19: cDNA cloning by functional complementation, mutation analysis in a patient with Zellweger syndrome, and potential role in peroxisomal membrane assembly.

Authors:  Y Matsuzono; N Kinoshita; S Tamura; N Shimozawa; M Hamasaki; K Ghaedi; R J Wanders; Y Suzuki; N Kondo; Y Fujiki
Journal:  Proc Natl Acad Sci U S A       Date:  1999-03-02       Impact factor: 11.205

5.  Peroxisome targeting signal type 1 (PTS1) receptor is involved in import of both PTS1 and PTS2: studies with PEX5-defective CHO cell mutants.

Authors:  H Otera; K Okumoto; K Tateishi; Y Ikoma; E Matsuda; M Nishimura; T Tsukamoto; T Osumi; K Ohashi; O Higuchi; Y Fujiki
Journal:  Mol Cell Biol       Date:  1998-01       Impact factor: 4.272

6.  The peroxin pex3p initiates membrane assembly in peroxisome biogenesis.

Authors:  K Ghaedi; S Tamura; K Okumoto; Y Matsuzono; Y Fujiki
Journal:  Mol Biol Cell       Date:  2000-06       Impact factor: 4.138

7.  PEX3 is the causal gene responsible for peroxisome membrane assembly-defective Zellweger syndrome of complementation group G.

Authors:  K Ghaedi; M Honsho; N Shimozawa; Y Suzuki; N Kondo; Y Fujiki
Journal:  Am J Hum Genet       Date:  2000-08-31       Impact factor: 11.025

8.  Peroxisome assembly factor 1: nonsense mutation in a peroxisome-deficient Chinese hamster ovary cell mutant and deletion analysis.

Authors:  T Tsukamoto; N Shimozawa; Y Fujiki
Journal:  Mol Cell Biol       Date:  1994-08       Impact factor: 4.272

9.  Mutations in novel peroxin gene PEX26 that cause peroxisome-biogenesis disorders of complementation group 8 provide a genotype-phenotype correlation.

Authors:  Naomi Matsumoto; Shigehiko Tamura; Satomi Furuki; Non Miyata; Ann Moser; Nobuyuki Shimozawa; Hugo W Moser; Yasuyuki Suzuki; Naomi Kondo; Yukio Fujiki
Journal:  Am J Hum Genet       Date:  2003-07-08       Impact factor: 11.025

10.  Mutation in PEX16 is causal in the peroxisome-deficient Zellweger syndrome of complementation group D.

Authors:  M Honsho; S Tamura; N Shimozawa; Y Suzuki; N Kondo; Y Fujiki
Journal:  Am J Hum Genet       Date:  1998-12       Impact factor: 11.025

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