Literature DB >> 18385758

Potential of fibroblast cell therapy for recessive dystrophic epidermolysis bullosa.

Tracy Wong1, Luke Gammon, Lu Liu, Jemima E Mellerio, Patricia J C Dopping-Hepenstal, John Pacy, George Elia, Rosemary Jeffery, Irene M Leigh, Harshad Navsaria, John A McGrath.   

Abstract

Recessive dystrophic epidermolysis bullosa (RDEB) is a severe inherited skin-blistering disorder caused by mutations in the COL7A1 gene that lead to reduced type-VII collagen and defective anchoring fibrils at the dermal-epidermal junction (DEJ). Presently there are no effective treatments for this disorder. Recent mouse studies have shown that intradermal injections of normal human fibroblasts can generate new human type-VII collagen and anchoring fibrils at the DEJ. To assess potential clinical benefits in humans, we gave single intradermal injections of allogeneic fibroblasts to five subjects with RDEB. We noted increased type-VII collagen at the DEJ at 2 weeks and at 3 months following injection and increased anchoring fibrils, although none of these had normal morphology. No adverse effects, clinical or immunopathologic, were noted. We believe the major effect of allogeneic fibroblasts is to increase the recipients' own COL7A1 mRNA levels with greater deposition of mutant type-VII collagen at the DEJ and formation of additional rudimentary anchoring fibrils. Nevertheless, this mutant protein may be partially functional and capable of increasing adhesion at the DEJ. This is the first study demonstrating that intradermal injections of allogeneic fibroblasts have therapeutic potential in human subjects with RDEB.

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Year:  2008        PMID: 18385758     DOI: 10.1038/jid.2008.78

Source DB:  PubMed          Journal:  J Invest Dermatol        ISSN: 0022-202X            Impact factor:   8.551


  71 in total

1.  Early intra-amniotic gene transfer using lentiviral vector improves skin blistering phenotype in a murine model of Herlitz junctional epidermolysis bullosa.

Authors:  M Endo; P W Zoltick; A Radu; Q Jiang; J Qiujie; C Matsui; P M Marinkovich; J McGrath; K Tamai; J Uitto; A W Flake
Journal:  Gene Ther       Date:  2011-09-22       Impact factor: 5.250

2.  From Mesoderm to Mesodermatology: Bone Marrow Mesenchymal Cells Heal Skin Wounds.

Authors:  Marketa Tolarova; Jakub Tolar
Journal:  Mol Ther       Date:  2015-08       Impact factor: 11.454

Review 3.  Regenerative medicine: Current therapies and future directions.

Authors:  Angelo S Mao; David J Mooney
Journal:  Proc Natl Acad Sci U S A       Date:  2015-11-24       Impact factor: 11.205

4.  Aminoglycosides restore full-length type VII collagen by overcoming premature termination codons: therapeutic implications for dystrophic epidermolysis bullosa.

Authors:  Jon Cogan; Jacqueline Weinstein; Xinyi Wang; Yingping Hou; Sabrina Martin; Andrew P South; David T Woodley; Mei Chen
Journal:  Mol Ther       Date:  2014-07-23       Impact factor: 11.454

5.  Can type VII collagen injections cure dystrophic epidermolysis bullosa?

Authors:  Leena Bruckner-Tuderman
Journal:  Mol Ther       Date:  2009-01       Impact factor: 11.454

Review 6.  [Epidermolysis bullosa. An update].

Authors:  H Schumann
Journal:  Hautarzt       Date:  2009-08       Impact factor: 0.751

Review 7.  [Orogenital and conjunctival involvement in hereditary and autoimmune blistering diseases].

Authors:  M Laimer; C M Lanschützer; M Emberger; E Nischler; J Stoiber; H Hintner
Journal:  Hautarzt       Date:  2009-11       Impact factor: 0.751

8.  Mechanisms of fibroblast cell therapy for dystrophic epidermolysis bullosa: high stability of collagen VII favors long-term skin integrity.

Authors:  Johannes S Kern; Stefan Loeckermann; Anja Fritsch; Ingrid Hausser; Wera Roth; Thomas M Magin; Claudia Mack; Marcel L Müller; Oliver Paul; Patrick Ruther; Leena Bruckner-Tuderman
Journal:  Mol Ther       Date:  2009-06-30       Impact factor: 11.454

Review 9.  Allogeneic blood and bone marrow cells for the treatment of severe epidermolysis bullosa: repair of the extracellular matrix.

Authors:  Jakub Tolar; John E Wagner
Journal:  Lancet       Date:  2013-10-05       Impact factor: 79.321

Review 10.  Modulating the stem cell niche for tissue regeneration.

Authors:  Steven W Lane; David A Williams; Fiona M Watt
Journal:  Nat Biotechnol       Date:  2014-08       Impact factor: 54.908

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