TY - JOUR
T1 - Predominance of Staphylococcus Correlates with Wound Burden and Disease Activity in Dystrophic Epidermolysis Bullosa
T2 - A Prospective Case-Control Study
AU - Reimer-Taschenbrecker, Antonia
AU - Künstner, Axel
AU - Hirose, Misa
AU - Hübner, Stefanie
AU - Gewert, Stella
AU - Ibrahim, Saleh
AU - Busch, Hauke
AU - Has, Cristina
N1 - Funding Information:
We thank Gabriele Grüninger and Aurelia Winter (University of Freiburg, Freiburg, Germany) and Miriam Freitag (University of Lübeck, Lübeck, Germany) for excellent technical assistance. This study was funded through a grant issued by DEBRA International and Austria to ART and CH . ART was supported by the Berta-Ottenstein-Programme, Faculty of Medicine, University of Freiburg . HB and AK acknowledge computational support from the OMICS compute cluster at the University of Lübeck. HB acknowledges funding by the Deutsche Forschungsgemeinschaft (German Research Foundation ) under Germany’s Excellence Strategy—EXC 22167-390884018 to HB. We kindly thank the patient organization Interessengemeinschaft Epidermolysis Bullosa e. V. DEBRA Deutschland for allowing us to join their family meeting, present this study, and collect samples. This study would not have been possible without the support of the participants, both with and without recessive dystrophic epidermolysis bullosa, and their families.
Funding Information:
We thank Gabriele Grüninger and Aurelia Winter (University of Freiburg, Freiburg, Germany) and Miriam Freitag (University of Lübeck, Lübeck, Germany) for excellent technical assistance. This study was funded through a grant issued by DEBRA International and Austria to ART and CH. ART was supported by the Berta-Ottenstein-Programme, Faculty of Medicine, University of Freiburg. HB and AK acknowledge computational support from the OMICS compute cluster at the University of Lübeck. HB acknowledges funding by the Deutsche Forschungsgemeinschaft (German Research Foundation) under Germany's Excellence Strategy—EXC 22167-390884018 to HB. We kindly thank the patient organization Interessengemeinschaft Epidermolysis Bullosa e. V. DEBRA Deutschland for allowing us to join their family meeting, present this study, and collect samples. This study would not have been possible without the support of the participants, both with and without recessive dystrophic epidermolysis bullosa, and their families. Conceptualization: ART, CH; Data Curation: ART, AK, HB, CH; Formal Analysis: CH; Funding Acquisition: ART, CH; Investigation: ART, MH, SH, SG; Methodology: MH, SI, HB, AK; Project Administration: ART, CH; Resources: ART, CH, MH, SI, AK, HB; Software: AK, HB; Supervision: HB, CH; Validation: ART, AK; Visualization: AK; Writing – Original Draft Preparation: ART, CH; Writing – Review and Editing: MH, SI, SH, SG, AK, HB
Publisher Copyright:
© 2022 The Authors
PY - 2022/8
Y1 - 2022/8
N2 - Recessive dystrophic epidermolysis bullosa is characterized by skin blistering and wounds. To uncover the changes in the skin and mucosal microbiome related to age and disease progression and microbiome impact on clinical and inflammatory laboratory parameters, swabs from wounded and unwounded skin, oral mucosa, and stool samples of 28 children with recessive dystrophic epidermolysis bullosa and 28 healthy controls were subjected to 16S-ribosomal RNA gene sequencing. Skin microbiome of patients with recessive dystrophic epidermolysis bullosa showed significantly reduced alpha diversity compared with that of healthy controls and showed significantly early, age-dependent predominance of Staphylococcus aureus, first in wounded skin and then in unwounded skin. These findings were more pronounced in the severe disease with higher abundances of S. aureus than in intermediate disease. S. aureus abundance correlated significantly with both acute and chronic wound burden. Changes in oral mucosal and gut microbiome were discrete, with no significant differences in alpha diversity. Our findings show that children with recessive dystrophic epidermolysis bullosa experience skin microbiome changes early in life. Longitudinal studies should confirm that dysbiosis starts in wounds and later extends to unwounded skin. The predominance of S. aureus significantly correlates with wound burden and disease activity and, to some extent, with systemic inflammation.
AB - Recessive dystrophic epidermolysis bullosa is characterized by skin blistering and wounds. To uncover the changes in the skin and mucosal microbiome related to age and disease progression and microbiome impact on clinical and inflammatory laboratory parameters, swabs from wounded and unwounded skin, oral mucosa, and stool samples of 28 children with recessive dystrophic epidermolysis bullosa and 28 healthy controls were subjected to 16S-ribosomal RNA gene sequencing. Skin microbiome of patients with recessive dystrophic epidermolysis bullosa showed significantly reduced alpha diversity compared with that of healthy controls and showed significantly early, age-dependent predominance of Staphylococcus aureus, first in wounded skin and then in unwounded skin. These findings were more pronounced in the severe disease with higher abundances of S. aureus than in intermediate disease. S. aureus abundance correlated significantly with both acute and chronic wound burden. Changes in oral mucosal and gut microbiome were discrete, with no significant differences in alpha diversity. Our findings show that children with recessive dystrophic epidermolysis bullosa experience skin microbiome changes early in life. Longitudinal studies should confirm that dysbiosis starts in wounds and later extends to unwounded skin. The predominance of S. aureus significantly correlates with wound burden and disease activity and, to some extent, with systemic inflammation.
UR - http://www.scopus.com/inward/record.url?scp=85126600690&partnerID=8YFLogxK
U2 - 10.1016/j.jid.2022.01.020
DO - 10.1016/j.jid.2022.01.020
M3 - Article
C2 - 35149000
AN - SCOPUS:85126600690
SN - 0022-202X
VL - 142
SP - 2117-2127.e8
JO - Journal of Investigative Dermatology
JF - Journal of Investigative Dermatology
IS - 8
ER -