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Título: Oral phenotype and salivary microbiome of individuals with Papillon–Lefèvre Syndrome
Autor(es): Lettieri, Giulia Melo
Santiago, Luander Medrado
Lettieri, Giancarlo Crosara
Borges, Luiz Gustavo dos Anjos
Marconatto, Letícia
Oliveira, Laudimar Alves de
Teixeira, Nailê Damé
Salles, Loise Pedrosa
ORCID: https://orcid.org/0000-0001-9369-8740
https://orcid.org/0000-0003-0165-0681
https://orcid.org/0000-0002-9300-1242
https://orcid.org/0000-0002-8680-2375
https://orcid.org/0000-0002-5578-5920
https://orcid.org/0000-0001-7959-7150
https://orcid.org/0000-0002-0490-0036
https://orcid.org/0000-0003-1061-7963
Afiliação do autor: University of Brasilia, Faculty of Health Sciences, Department of Dentistry
University of Brasilia, Faculty of Health Sciences, Department of Dentistry
Specialized Center in Periodontology and Implantology, Periodontology Research Group
Helmholtz Centre for Infection Research, Microbial Interactions and Processes Research Group, Braunschweig, Germany
Pontifical Catholic University of Rio Grande do Sul, Institute of Petroleum and Natural Resources
Helmholtz Centre for Infection Research, Microbial Interactions and Processes Research Group, Braunschweig, Germany
University of Brasilia, Faculty of Health Sciences, Department of Dentistry
University of Brasilia, Faculty of Health Sciences, Department of Dentistry
University of Brasilia, Faculty of Health Sciences, Department of Dentistry
Assunto: Periodontite
Microbiologia
Dentes - doenças
Catepsina C
Data de publicação: 26-Ago-2021
Editora: Frontiers
Referência: LETTIERI, Giulia Melo et. al. Oral phenotype and salivary microbiome of individuals with Papillon–Lefèvre Syndrome. Frontiers in Cellular and Infection Microbiology, [S.l.], v. 11, 2021. DOI: https://doi.org/10.3389/fcimb.2021.720790. Disponível em: https://www.frontiersin.org/journals/cellular-and-infection-microbiology/articles/10.3389/fcimb.2021.720790/full. Acesso em: 13 mar. 2025.
Abstract: Papillon–Lefèvre syndrome (PLS) is an autosomal recessive rare disease, main characteristics of which include palmoplantar hyperkeratosis and premature edentulism due to advanced periodontitis (formerly aggressive periodontitis). This study aimed to characterize the oral phenotype, including salivary parameters, and the salivary microbiome of three PLS sisters, comparatively. Two sisters were toothless (PLSTL1 and PLSTL2), and one sister had most of the teeth in the oral cavity (PLST). Total DNA was extracted from the unstimulated saliva, and the amplicon sequencing of the 16S rRNA gene fragment was performed in an Ion PGM platform. The amplicon sequence variants (ASVs) were obtained using the DADA2 pipeline, and the taxonomy was assigned using the SILVA v.138. The main phenotypic characteristics of PLS were bone loss and premature loss of primary and permanent dentition. The PLST sister presented advanced periodontitis with gingival bleeding and suppuration, corresponding to the advanced periodontitis as a manifestation of systemic disease, stage IV, grade C. All three PLS sisters presented hyposalivation as a possible secondary outcome of the syndrome. Interestingly, PLST salivary microbiota was dominated by the uncultured bacteria Bacterioidales (F0058), Fusobacterium, Treponema, and Sulfophobococcus (Archaea domain). Streptococcus, Haemophilus, and Caldivirga (Archaea) dominated the microbiome of the PLSTL1 sister, while the PLSTL2 had higher abundances of Lactobacillus and Porphyromonas. This study was the first to show a high abundance of organisms belonging to the Archaea domain comprising a core microbiome in human saliva. In conclusion, a PLST individual does have a microbiota different from that of the periodontitis’ aggressiveness previously recognized. Due to an ineffective cathepsin C, the impairment of neutrophils probably provided a favorable environment for the PLS microbiome. The interactions of Bacteroidales F0058, Caldivirga, and Sulfophobococcus with the microbial consortium of PLS deserves future investigation. Traditional periodontal therapy is not efficient in PLS patients. Unraveling the PLS microbiome is essential in searching for appropriate treatment and avoiding early tooth loss.
Unidade Acadêmica: Faculdade de Ciências da Saúde (FS)
Departamento de Odontologia (FS ODT)
Programa de pós-graduação: Programa de Pós-Graduação em Odontologia
Licença: This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
DOI: https://doi.org/10.3389/fcimb.2021.720790
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