Literature DB >> 28357176

Novel and emerging therapies in the treatment of recessive dystrophic epidermolysis bullosa.

Ellie Rashidghamat1, John A McGrath1.   

Abstract

Epidermolysis bullosa (EB) is a clinically and genetically heterogeneous group of inherited blistering diseases that affects ∼ 500,000 people worldwide. Clinically, individuals with EB have fragile skin and are susceptible to blistering following minimal trauma, with mucous membrane and other organ involvement in some subtypes. Within the spectrum of EB, ∼ 5% of affected individuals have the clinically more severe recessive dystrophic (RDEB) variant with a prevalence of 8 per one million of the population. RDEB is caused by loss-of-function mutations in the type VII collagen gene, COL7A1, which leads to reduced or absent type VII collagen (C7) and a paucity of structurally effective anchoring fibrils at the dermal-epidermal junction (DEJ). Currently, there is no cure for RDEB, although considerable progress has been made in testing novel treatments including gene therapy (lentiviral and gamma retroviral vectors for COL7A1 supplementation in keratinocytes and fibroblasts), as well as cell therapy (use of allogeneic fibroblasts, mesenchymal stromal cells (MSCs), and bone marrow transplantation (BMT)). Here, we review current treatment modalities available as well as novel and emerging therapies in the treatment of RDEB. Clinical trials of new translational therapies in RDEB offer hope for improved clinical management of patients as well as generating broader lessons for regenerative medicine that could be applicable to other inherited or acquired abnormalities of wound healing or scarring.

Entities:  

Keywords:  Epidermolysis bullosa; cell; gene; protein; treatment

Year:  2017        PMID: 28357176      PMCID: PMC5359356          DOI: 10.5582/irdr.2017.01005

Source DB:  PubMed          Journal:  Intractable Rare Dis Res        ISSN: 2186-3644


  144 in total

1.  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

2.  HB-EGF induces COL7A1 expression in keratinocytes and fibroblasts: possible mechanism underlying allogeneic fibroblast therapy in recessive dystrophic epidermolysis Bullosa.

Authors:  Nikoletta Nagy; Noor Almaani; Akio Tanaka; Joey E Lai-Cheong; Tanasit Techanukul; Jemima E Mellerio; John A McGrath
Journal:  J Invest Dermatol       Date:  2011-04-07       Impact factor: 8.551

3.  Targeted Exon Skipping Restores Type VII Collagen Expression and Anchoring Fibril Formation in an In Vivo RDEB Model.

Authors:  Sandrina Turczynski; Matthias Titeux; Laure Tonasso; Audrey Décha; Akemi Ishida-Yamamoto; Alain Hovnanian
Journal:  J Invest Dermatol       Date:  2016-08-03       Impact factor: 8.551

4.  Nutritional outcome in children with severe generalized recessive dystrophic epidermolysis bullosa: a short- and long-term evaluation of gastrostomy and enteral feeding.

Authors:  V Colomb; E Bourdon-Lannoy; C Lambe; F Sauvat; S Hadj Rabia; D Teillac; Y De Prost; C Bodemer
Journal:  Br J Dermatol       Date:  2012-02       Impact factor: 9.302

Review 5.  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

6.  Progenitor cell trafficking is regulated by hypoxic gradients through HIF-1 induction of SDF-1.

Authors:  Daniel J Ceradini; Anita R Kulkarni; Matthew J Callaghan; Oren M Tepper; Nicholas Bastidas; Mark E Kleinman; Jennifer M Capla; Robert D Galiano; Jamie P Levine; Geoffrey C Gurtner
Journal:  Nat Med       Date:  2004-07-04       Impact factor: 53.440

Review 7.  Management of cutaneous squamous cell carcinoma in patients with epidermolysis bullosa: best clinical practice guidelines.

Authors:  J E Mellerio; S J Robertson; C Bernardis; A Diem; J D Fine; R George; D Goldberg; G B Halmos; M Harries; M F Jonkman; A Lucky; A E Martinez; E Maubec; S Morris; D F Murrell; F Palisson; E I Pillay; A Robson; J C Salas-Alanis; J A McGrath
Journal:  Br J Dermatol       Date:  2015-11-07       Impact factor: 9.302

8.  Normal and gene-corrected dystrophic epidermolysis bullosa fibroblasts alone can produce type VII collagen at the basement membrane zone.

Authors:  David T Woodley; Gerald G Krueger; Cynthia M Jorgensen; Janet A Fairley; Tom Atha; Yi Huang; Lawrence Chan; Douglas R Keene; Mei Chen
Journal:  J Invest Dermatol       Date:  2003-11       Impact factor: 8.551

Review 9.  Type VII Collagen Replacement Therapy in Recessive Dystrophic Epidermolysis Bullosa-How Much, How Often?

Authors:  Andrew P South; Jouni Uitto
Journal:  J Invest Dermatol       Date:  2016-06       Impact factor: 8.551

10.  A Gene Gun-mediated Nonviral RNA trans-splicing Strategy for Col7a1 Repair.

Authors:  Patricia Peking; Ulrich Koller; Stefan Hainzl; Sophie Kitzmueller; Thomas Kocher; Elisabeth Mayr; Alexander Nyström; Thomas Lener; Julia Reichelt; Johann W Bauer; Eva M Murauer
Journal:  Mol Ther Nucleic Acids       Date:  2016-03-01       Impact factor: 10.183

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  24 in total

1.  Previously Unreported COL7A1 Mutation in a Somali Patient with Dystrophic Epidermolysis Bullosa.

Authors:  Valeria Venti; Bruna Scalia; Alessandra Sauna; Maria Rita Nasca; Pierluigi Smilari; Andrea D Praticò; Agata Fiumara; Xena G Pappalardo; Piero Pavone
Journal:  Mol Syndromol       Date:  2019-11-16

2.  CRISPR/Cas9-based targeted genome editing for correction of recessive dystrophic epidermolysis bullosa using iPS cells.

Authors:  Joanna Jacków; Zongyou Guo; Corey Hansen; Hasan E Abaci; Yanne S Doucet; Jung U Shin; Ryota Hayashi; Dominick DeLorenzo; Yudai Kabata; Satoru Shinkuma; Julio C Salas-Alanis; Angela M Christiano
Journal:  Proc Natl Acad Sci U S A       Date:  2019-12-09       Impact factor: 11.205

3.  Interrogation of RDEB Epidermal Allografts after BMT Reveals Coexpression of Collagen VII and Keratin 15 with Proinflammatory Immune Cells and Fibroblasts.

Authors:  Julia A Riedl; Megan Riddle; Lily Xia; Cindy Eide; Christina Boull; Christen L Ebens; Jakub Tolar
Journal:  J Invest Dermatol       Date:  2022-03-15       Impact factor: 7.590

Review 4.  Bullous Diseases in Children: A Review of Clinical Features and Treatment Options.

Authors:  Brittney Schultz; Kristen Hook
Journal:  Paediatr Drugs       Date:  2019-10       Impact factor: 3.022

5.  ABCB5+ dermal mesenchymal stromal cells with favorable skin homing and local immunomodulation for recessive dystrophic epidermolysis bullosa treatment.

Authors:  Julia Riedl; Michael Pickett-Leonard; Cindy Eide; Mark Andreas Kluth; Christoph Ganss; Natasha Y Frank; Markus H Frank; Christen L Ebens; Jakub Tolar
Journal:  Stem Cells       Date:  2021-03-01       Impact factor: 5.845

6.  COL7A1 Editing via CRISPR/Cas9 in Recessive Dystrophic Epidermolysis Bullosa.

Authors:  Stefan Hainzl; Patricia Peking; Thomas Kocher; Eva M Murauer; Fernando Larcher; Marcela Del Rio; Blanca Duarte; Markus Steiner; Alfred Klausegger; Johann W Bauer; Julia Reichelt; Ulrich Koller
Journal:  Mol Ther       Date:  2017-07-13       Impact factor: 11.454

Review 7.  A rare case of skin blistering and esophageal stenosis in the course of epidermolysis bullosa - case report and literature review.

Authors:  Agata Michalak; Halina Cichoż-Lach; Beata Prozorow-Król; Leszek Buk; Monika Dzida
Journal:  BMC Gastroenterol       Date:  2018-04-13       Impact factor: 3.067

Review 8.  From Structure to Phenotype: Impact of Collagen Alterations on Human Health.

Authors:  Lavinia Arseni; Anita Lombardi; Donata Orioli
Journal:  Int J Mol Sci       Date:  2018-05-08       Impact factor: 5.923

Review 9.  Molecular Therapeutics in Development for Epidermolysis Bullosa: Update 2020.

Authors:  Cristina Has; Andrew South; Jouni Uitto
Journal:  Mol Diagn Ther       Date:  2020-06       Impact factor: 4.074

10.  Exposed CendR Domain in Homing Peptide Yields Skin-Targeted Therapeutic in Epidermolysis Bullosa.

Authors:  Toini Pemmari; Larisa Ivanova; Ulrike May; Prakash Lingasamy; Allan Tobi; Anja Pasternack; Stuart Prince; Olli Ritvos; Shreya Makkapati; Tambet Teesalu; Mitchell S Cairo; Tero A H Järvinen; Yanling Liao
Journal:  Mol Ther       Date:  2020-05-20       Impact factor: 11.454

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