Literature DB >> 2158095

Restoration of normal lysosomal function in mucopolysaccharidosis type VII cells by retroviral vector-mediated gene transfer.

J H Wolfe1, E H Schuchman, L E Stramm, E A Concaugh, M E Haskins, G D Aguirre, D F Patterson, R J Desnick, E Gilboa.   

Abstract

Retroviral vectors were constructed containing a rat beta-glucuronidase cDNA driven by heterologous promoters. Vector-mediated gene transfer into human and canine beta-glucuronidase-deficient mucopolysaccharidosis type VII fibroblasts completely corrected the deficiency in beta-glucuronidase enzymatic activity. In primary cultures of canine mucopolysaccharidosis type VII retinal pigment epithelial cells, which contain large amounts of undegraded glycosaminoglycan substrates, vector correction restored normal processing of specific glycosaminoglycans in the lysosomal compartment. In canine mucopolysaccharidosis type VII bone marrow cells, beta-glucuronidase was expressed at high levels in transduced cells. Thus, the vector-encoded beta-glucuronidase was expressed at therapeutic levels in the appropriate organelle and corrected the metabolic defect in cells exhibiting the characteristic pathology of this lysosomal storage disorder.

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Year:  1990        PMID: 2158095      PMCID: PMC53796          DOI: 10.1073/pnas.87.8.2877

Source DB:  PubMed          Journal:  Proc Natl Acad Sci U S A        ISSN: 0027-8424            Impact factor:   11.205


  40 in total

1.  Cloning, sequencing, and expression of cDNA for human beta-glucuronidase.

Authors:  A Oshima; J W Kyle; R D Miller; J W Hoffmann; P P Powell; J H Grubb; W S Sly; M Tropak; K S Guise; R A Gravel
Journal:  Proc Natl Acad Sci U S A       Date:  1987-02       Impact factor: 11.205

2.  Redesign of retrovirus packaging cell lines to avoid recombination leading to helper virus production.

Authors:  A D Miller; C Buttimore
Journal:  Mol Cell Biol       Date:  1986-08       Impact factor: 4.272

3.  Expression of a foreign gene in myeloid and lymphoid cells derived from multipotent haematopoietic precursors.

Authors:  G Keller; C Paige; E Gilboa; E F Wagner
Journal:  Nature       Date:  1985 Nov 14-20       Impact factor: 49.962

4.  Construction of a retrovirus packaging mutant and its use to produce helper-free defective retrovirus.

Authors:  R Mann; R C Mulligan; D Baltimore
Journal:  Cell       Date:  1983-05       Impact factor: 41.582

5.  Beta-glucuronidase deficiency in a dog: a model of human mucopolysaccharidosis VII.

Authors:  M E Haskins; R J Desnick; N DiFerrante; P F Jezyk; D F Patterson
Journal:  Pediatr Res       Date:  1984-10       Impact factor: 3.756

6.  Retrovirus-mediated transfer of the human glucocerebrosidase gene to Gaucher fibroblasts.

Authors:  P V Choudary; J A Barranger; S Tsuji; J Mayor; M E LaMarca; C L Cepko; R C Mulligan; E I Ginns
Journal:  Mol Biol Med       Date:  1986-06

7.  Nucleotide sequence of rat preputial gland beta-glucuronidase cDNA and in vitro insertion of its encoded polypeptide into microsomal membranes.

Authors:  Y Nishimura; M G Rosenfeld; G Kreibich; U Gubler; D D Sabatini; M Adesnik; R Andy
Journal:  Proc Natl Acad Sci U S A       Date:  1986-10       Impact factor: 11.205

8.  Efficient retrovirus-mediated transfer and expression of a human adenosine deaminase gene in diploid skin fibroblasts from an adenosine deaminase-deficient human.

Authors:  T D Palmer; R A Hock; W R Osborne; A D Miller
Journal:  Proc Natl Acad Sci U S A       Date:  1987-02       Impact factor: 11.205

9.  Correction of adenosine deaminase deficiency in cultured human T and B cells by retrovirus-mediated gene transfer.

Authors:  P W Kantoff; D B Kohn; H Mitsuya; D Armentano; M Sieberg; J A Zwiebel; M A Eglitis; J R McLachlin; D A Wiginton; J J Hutton
Journal:  Proc Natl Acad Sci U S A       Date:  1986-09       Impact factor: 11.205

10.  Retroviral transfer of genes into canine hemopoietic progenitor cells in culture: a model for human gene therapy.

Authors:  W W Kwok; F Schuening; R B Stead; A D Miller
Journal:  Proc Natl Acad Sci U S A       Date:  1986-06       Impact factor: 11.205

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  8 in total

1.  Restoration of arylsulphatase A activity in human-metachromatic-leucodystrophy fibroblasts via retroviral-vector-mediated gene transfer.

Authors:  W Rommerskirch; A L Fluharty; C Peters; K von Figura; V Gieselmann
Journal:  Biochem J       Date:  1991-12-01       Impact factor: 3.857

2.  Possible beta-mannosidosis chimera. Altered expression of metabolic perturbations.

Authors:  M Z Jones; K T Cavanagh; R Kranich; C Traviss; Y Fujita; M Ohta; F Matsuura
Journal:  J Inherit Metab Dis       Date:  1993       Impact factor: 4.982

3.  Long-term in vitro correction of alpha-L-iduronidase deficiency (Hurler syndrome) in human bone marrow.

Authors:  L J Fairbairn; L S Lashford; E Spooncer; R H McDermott; G Lebens; J E Arrand; J R Arrand; I Bellantuono; R Holt; C E Hatton; A Cooper; G T Besley; J E Wraith; D S Anson; J J Hopwood; T M Dexter
Journal:  Proc Natl Acad Sci U S A       Date:  1996-03-05       Impact factor: 11.205

4.  Genes transferred by retroviral vectors into normal and mutant myoblasts in primary cultures are expressed in myotubes.

Authors:  B F Smith; R K Hoffman; U Giger; J H Wolfe
Journal:  Mol Cell Biol       Date:  1990-06       Impact factor: 4.272

5.  A single-base-pair deletion in the beta-glucuronidase gene accounts for the phenotype of murine mucopolysaccharidosis type VII.

Authors:  M S Sands; E H Birkenmeier
Journal:  Proc Natl Acad Sci U S A       Date:  1993-07-15       Impact factor: 11.205

6.  Enzyme replacement therapy for murine mucopolysaccharidosis type VII.

Authors:  M S Sands; C Vogler; J W Kyle; J H Grubb; B Levy; N Galvin; W S Sly; E H Birkenmeier
Journal:  J Clin Invest       Date:  1994-06       Impact factor: 14.808

7.  Mucopolysaccharidosis type VII (Sly syndrome). Beta-glucuronidase-deficient mucopolysaccharidosis in the dog.

Authors:  M E Haskins; G D Aguirre; P F Jezyk; E H Schuchman; R J Desnick; D F Patterson
Journal:  Am J Pathol       Date:  1991-06       Impact factor: 4.307

8.  Restoration of arylsulphatase B activity in human mucopolysaccharidosis-type-VI fibroblasts by retroviral-vector-mediated gene transfer.

Authors:  C Peters; W Rommerskirch; S Modaressi; K von Figura
Journal:  Biochem J       Date:  1991-06-01       Impact factor: 3.857

  8 in total

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