Impact of low-dose calcipotriol ointment on wound healing, pruritus and pain in patients with dystrophic epidermolysis bullosa: A randomized, double-blind, placebo-controlled trial.


Journal

Orphanet journal of rare diseases
ISSN: 1750-1172
Titre abrégé: Orphanet J Rare Dis
Pays: England
ID NLM: 101266602

Informations de publication

Date de publication:
08 11 2021
Historique:
received: 17 05 2021
accepted: 30 09 2021
entrez: 9 11 2021
pubmed: 10 11 2021
medline: 12 11 2021
Statut: epublish

Résumé

Wound management is a critical factor when treating patients with the inherited skin fragility disease dystrophic epidermolysis bullosa (DEB). Due to genetic defects in structural proteins, skin and mucous epithelia are prone to blistering and chronic wounding upon minor trauma. Furthermore, these wounds are commonly associated with excessive pruritus and predispose to the development of life-threatening squamous cell carcinomas, underscoring the unmet need for new therapeutic options to improve wound healing in this patient cohort. Vitamin D3 is acknowledged to play an important role in wound healing by modulating different cellular processes that impact epidermal homeostasis and immune responses. In this study, we evaluate the safety and efficacy of low-dose calcipotriol, a vitamin D3 analogue, in promoting wound healing and reducing itch and pain in patients with DEB. Eligible DEB patients, aged ≥ 6 years and with a known mutation in the COL7A1 gene, were recruited to a placebo-controlled, randomized, double blind, cross-over phase II monocentric clinical trial. Patients were required to have at least two wounds with a minimum size of 6 cm Six patients completed the clinical trial and were included into the final analysis. Topical low-dose calcipotriol treatment led to a significant reduction in wound area at day 14 compared to placebo (88.4% vs. 65.5%, P < 0.05). Patients also reported a significant reduction of pruritus with calcipotriol ointment compared to placebo over the entire course of the treatment as shown by itch scores of 3.16 vs 4.83 (P < 0.05) and 1.83 vs 5.52 (P < 0.0001) at days 14 and 28, respectively. Treatment with low-dose calcipotriol did not affect serum calcium levels and improved the species richness of the wound microbiome, albeit with no statistical significance. Our results show that topical treatment with low-dose calcipotriol can accelerate wound closure and significantly reduces itch, and can be considered a safe and readily-available option to improve local wound care in DEB patients. Trial Registration EudraCT: 2016-001,967-35. Registered 28 June 2016, https://www.clinicaltrialsregister.eu/ctr-search/trial/2016-001967-35/AT.

Sections du résumé

BACKGROUND
Wound management is a critical factor when treating patients with the inherited skin fragility disease dystrophic epidermolysis bullosa (DEB). Due to genetic defects in structural proteins, skin and mucous epithelia are prone to blistering and chronic wounding upon minor trauma. Furthermore, these wounds are commonly associated with excessive pruritus and predispose to the development of life-threatening squamous cell carcinomas, underscoring the unmet need for new therapeutic options to improve wound healing in this patient cohort. Vitamin D3 is acknowledged to play an important role in wound healing by modulating different cellular processes that impact epidermal homeostasis and immune responses. In this study, we evaluate the safety and efficacy of low-dose calcipotriol, a vitamin D3 analogue, in promoting wound healing and reducing itch and pain in patients with DEB.
METHODS
Eligible DEB patients, aged ≥ 6 years and with a known mutation in the COL7A1 gene, were recruited to a placebo-controlled, randomized, double blind, cross-over phase II monocentric clinical trial. Patients were required to have at least two wounds with a minimum size of 6 cm
RESULTS
Six patients completed the clinical trial and were included into the final analysis. Topical low-dose calcipotriol treatment led to a significant reduction in wound area at day 14 compared to placebo (88.4% vs. 65.5%, P < 0.05). Patients also reported a significant reduction of pruritus with calcipotriol ointment compared to placebo over the entire course of the treatment as shown by itch scores of 3.16 vs 4.83 (P < 0.05) and 1.83 vs 5.52 (P < 0.0001) at days 14 and 28, respectively. Treatment with low-dose calcipotriol did not affect serum calcium levels and improved the species richness of the wound microbiome, albeit with no statistical significance.
CONCLUSIONS
Our results show that topical treatment with low-dose calcipotriol can accelerate wound closure and significantly reduces itch, and can be considered a safe and readily-available option to improve local wound care in DEB patients. Trial Registration EudraCT: 2016-001,967-35. Registered 28 June 2016, https://www.clinicaltrialsregister.eu/ctr-search/trial/2016-001967-35/AT.

Identifiants

pubmed: 34749770
doi: 10.1186/s13023-021-02062-2
pii: 10.1186/s13023-021-02062-2
pmc: PMC8576995
doi:

Substances chimiques

COL7A1 protein, human 0
Collagen Type VII 0
Ointments 0
calcipotriene 143NQ3779B
Calcitriol FXC9231JVH

Types de publication

Journal Article Randomized Controlled Trial Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

473

Informations de copyright

© 2021. The Author(s).

Références

J Eur Acad Dermatol Venereol. 2015 Feb;29(2):396-398
pubmed: 24533594
Br J Dermatol. 2013 Nov;169(5):1025-33
pubmed: 24032424
Sci Rep. 2020 Sep 15;10(1):15064
pubmed: 32934247
Medicine (Baltimore). 2015 Oct;94(42):e1528
pubmed: 26496255
J Dermatol. 2013 Jun;40(6):424-7
pubmed: 23414298
Acta Derm Venereol. 2021 Aug 24;101(8):adv00523
pubmed: 34230977
J Invest Dermatol. 2017 Nov;137(11):2298-2308
pubmed: 28736230
FASEB J. 2005 Jul;19(9):1067-77
pubmed: 15985530
Orphanet J Rare Dis. 2020 Jan 3;15(1):1
pubmed: 31900176
Orphanet J Rare Dis. 2021 Apr 13;16(1):175
pubmed: 33849616
Int J Mol Sci. 2017 Oct 18;18(10):
pubmed: 29057787
Br J Dermatol. 2020 Jun;182(6):1437-1448
pubmed: 31487386
Sci Rep. 2018 Sep 7;8(1):13430
pubmed: 30194425
Int Wound J. 2014 Feb;11(1):44-9
pubmed: 22776565
Nat Rev Endocrinol. 2020 Apr;16(4):234-252
pubmed: 32029884
Science. 2012 Aug 31;337(6098):1115-9
pubmed: 22837383
J Invest Dermatol. 2015 Nov;135(11):2887-2890
pubmed: 26121211
Cell Rep. 2019 Nov 26;29(9):2689-2701.e4
pubmed: 31775038
Nat Commun. 2015 Jan 09;6:5932
pubmed: 25575023
Acta Derm Venereol. 2018 Jan 12;98(1):70-76
pubmed: 28853495
J Autoimmun. 2017 Dec;85:78-97
pubmed: 28733125
JAMA Dermatol. 2016 Nov 1;152(11):1231-1238
pubmed: 27463098
J Invest Dermatol. 2018 Nov;138(11):2492-2495
pubmed: 29753707

Auteurs

Christina Guttmann-Gruber (C)

EB House Austria, Research Program for Molecular Therapy of Genodermatoses, Department of Dermatology and Allergology, University Hospital of the Paracelsus Medical University, Salzburg, Austria.

Josefina Piñón Hofbauer (J)

EB House Austria, Research Program for Molecular Therapy of Genodermatoses, Department of Dermatology and Allergology, University Hospital of the Paracelsus Medical University, Salzburg, Austria.

Birgit Tockner (B)

EB House Austria, Research Program for Molecular Therapy of Genodermatoses, Department of Dermatology and Allergology, University Hospital of the Paracelsus Medical University, Salzburg, Austria.

Victoria Reichl (V)

EB House Austria, Research Program for Molecular Therapy of Genodermatoses, Department of Dermatology and Allergology, University Hospital of the Paracelsus Medical University, Salzburg, Austria.

Alfred Klausegger (A)

EB House Austria, Research Program for Molecular Therapy of Genodermatoses, Department of Dermatology and Allergology, University Hospital of the Paracelsus Medical University, Salzburg, Austria.

Peter Hofbauer (P)

Landesapotheke Salzburg, Department of Production, Hospital Pharmacy, Salzburg, Austria.

Martin Wolkersdorfer (M)

Landesapotheke Salzburg, Department of Production, Hospital Pharmacy, Salzburg, Austria.

Khek-Chian Tham (KC)

Skin Research Institute of Singapore, A*STAR, 8A Biomedical Grove, Immunos #06-06, Singapore, Singapore.

Seong Soo Lim (SS)

Skin Research Institute of Singapore, A*STAR, 8A Biomedical Grove, Immunos #06-06, Singapore, Singapore.

John E Common (JE)

Skin Research Institute of Singapore, A*STAR, 8A Biomedical Grove, Immunos #06-06, Singapore, Singapore.

Anja Diem (A)

EB House Austria, Outpatient Unit, Department of Dermatology and Allergology, Paracelsus Medical University, Salzburg, Austria.

Katharina Ude-Schoder (K)

EB House Austria, Outpatient Unit, Department of Dermatology and Allergology, Paracelsus Medical University, Salzburg, Austria.

Wolfgang Hitzl (W)

Research Office Biostatistics, Paracelsus Medical University, Salzburg, Austria.
Department of Ophthalmology and Optometry, Paracelsus Medical University Salzburg, Müllner Hauptstr. 48, 5020, Salzburg, Austria.
Research Program Experimental Ophthalmology and Glaucoma Research, Paracelsus Medical University, Muellner Hauptstrasse 48, 5020, Salzburg, Austria.

Florian Lagler (F)

Institute for Inborn Errors of Metabolism and Department of Pediatrics, Paracelsus Medical University, Salzburg, Austria.

Julia Reichelt (J)

EB House Austria, Research Program for Molecular Therapy of Genodermatoses, Department of Dermatology and Allergology, University Hospital of the Paracelsus Medical University, Salzburg, Austria.

Johann W Bauer (JW)

EB House Austria, Research Program for Molecular Therapy of Genodermatoses, Department of Dermatology and Allergology, University Hospital of the Paracelsus Medical University, Salzburg, Austria.
Department of Dermatology and Allergology, University Hospital Salzburg, Paracelsus Medical University, Muellner-Hauptstrasse 48, 5020, Salzburg, Austria.

Roland Lang (R)

Department of Dermatology and Allergology, University Hospital Salzburg, Paracelsus Medical University, Muellner-Hauptstrasse 48, 5020, Salzburg, Austria. r.lang@salk.at.

Martin Laimer (M)

EB House Austria, Research Program for Molecular Therapy of Genodermatoses, Department of Dermatology and Allergology, University Hospital of the Paracelsus Medical University, Salzburg, Austria. m.laimer@salk.at.
Department of Dermatology and Allergology, University Hospital Salzburg, Paracelsus Medical University, Muellner-Hauptstrasse 48, 5020, Salzburg, Austria. m.laimer@salk.at.

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