Burosumab and Dental Abscesses in Children With X-Linked Hypophosphatemia.

BONE MATRIX CELL/TISSUE SIGNALING – ENDOCRINE PATHWAYS DENTAL BIOLOGY DISORDERS OF CALCIUM/PHOSPHATE METABOLISM PTH/VIT D/FGF23 |MATRIX MINERALIZATION

Journal

JBMR plus
ISSN: 2473-4039
Titre abrégé: JBMR Plus
Pays: England
ID NLM: 101707013

Informations de publication

Date de publication:
Nov 2022
Historique:
received: 10 05 2022
revised: 08 07 2022
accepted: 27 07 2022
entrez: 18 11 2022
pubmed: 19 11 2022
medline: 19 11 2022
Statut: epublish

Résumé

X-linked hypophosphatemia (XLH) is a rare genetic disorder that disrupts skeletal and dental mineralization. In addition to rickets in children, XLH patients also have frequent spontaneous dental abscesses that increase the risk of tooth loss and may lead to facial cellulitis. Hypomineralized and hypoplastic dentin is the main driver of these infections. Conventional treatment (CT) of XLH improves this tissue defect and reduces the occurrence of dental abscesses. Burosumab is a recent treatment for XLH that targets excess circulating fibroblast growth factor 23 (FGF23), and its benefits on rickets have been demonstrated. It is not yet known whether burosumab improves dental manifestations of XLH. The main objective of our study was to compare the incidence of dental abscesses with XLH treated with either CT or burosumab. In this monocentric retrospective study, we measured and compared the incidence of dental abscess in children with XLH treated with either CT or burosumab, followed at our dental center for at least 1 year. The primary endpoint was the number of dental abscesses per month of dental follow-up. A total of 71 children were included in the study, with a mean ± standard deviation (SD) age at the start of dental follow-up of 7.86 ± 3.76. Thirty-eight children were treated with CT (53.5%) and 33 with burosumab (46.5%). All children treated with burosumab had previously been treated with CT. The mean number of dental abscesses per month of dental follow-up was significantly reduced in the burosumab group compared with the CT group (0.01 versus 0.04;

Identifiants

pubmed: 36398111
doi: 10.1002/jbm4.10672
pii: JBM410672
pmc: PMC9664523
doi:

Types de publication

Journal Article

Langues

eng

Pagination

e10672

Informations de copyright

© 2022 The Authors. JBMR Plus published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research.

Déclaration de conflit d'intérêts

The authors declared the following potential conflicts of interest with respect to the research, authorship, and/or publication of this article: AL and MBD have received honoraria, grant for research in other projects independent of this study from Kyowa Kirin Pharma. CC has received grants for research in other projects independent of this study from Kyowa Kirin Pharma. AR and VSF have received honoraria from Kyowa Kirin Pharma. All other authors have no relevant financial or nonfinancial interests to disclose.

Références

PLoS One. 2013;8(2):e56749
pubmed: 23451077
Endocr Connect. 2014 Mar 14;3(1):R13-30
pubmed: 24550322
JBMR Plus. 2021 Mar 22;5(5):e10470
pubmed: 33977199
Pediatr Dent. 1991 Mar-Apr;13(2):91-6
pubmed: 1652746
Int J Paediatr Dent. 1998 Mar;8(1):19-28
pubmed: 9558542
J Dent Res. 2021 Dec;100(13):1482-1491
pubmed: 33906518
Clin Oral Investig. 2015 May;19(4):759-68
pubmed: 25677243
Orphanet J Rare Dis. 2019 Feb 26;14(1):58
pubmed: 30808384
Eur J Paediatr Dent. 2006 Jun;7(2):61-6
pubmed: 16842025
Calcif Tissue Int. 2006 Nov;79(5):294-300
pubmed: 17115324
J Clin Endocrinol Metab. 2022 Jul 14;107(8):e3241-e3253
pubmed: 35533340
Endocr Rev. 2014 Feb;35(1):1-34
pubmed: 23939820
Pediatr Infect Dis J. 2006 Apr;25(4):339-42
pubmed: 16567986
Endocr Connect. 2022 Jan 28;11(1):
pubmed: 34941571
Cells Tissues Organs. 2009;189(1-4):219-23
pubmed: 18701809
J Dent Res. 1963 Nov-Dec;42:1490-502
pubmed: 14081973
J Oral Rehabil. 2012 Feb;39(2):144-50
pubmed: 21902707
Nat Rev Nephrol. 2019 Jul;15(7):435-455
pubmed: 31068690
N Engl J Med. 2018 May 24;378(21):1987-1998
pubmed: 29791829
J Oral Rehabil. 2021 Feb;48(2):160-168
pubmed: 33058298
Lancet. 2019 Jun 15;393(10189):2416-2427
pubmed: 31104833
J Bone Miner Res. 2018 Aug;33(8):1383-1393
pubmed: 29947083
J Pediatr. 2003 Mar;142(3):324-31
pubmed: 12640383
J Endocr Soc. 2019 May 07;3(7):1321-1334
pubmed: 31259293
Front Endocrinol (Lausanne). 2018 May 02;9:189
pubmed: 29770125
J Dent Educ. 2003 Jun;67(6):678-89
pubmed: 12856968
Nat Rev Endocrinol. 2021 Jun;17(6):336-349
pubmed: 33948016
Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2009 Apr;107(4):525-30
pubmed: 19201216
J Dent Res. 2017 Apr;96(4):388-395
pubmed: 27821544
JBMR Plus. 2021 Mar 03;5(4):e10463
pubmed: 33869987
J Clin Invest. 2016 Mar 1;126(3):962-74
pubmed: 26878171
Oral Dis. 2007 Sep;13(5):482-9
pubmed: 17714351
J Am Soc Nephrol. 2016 Jan;27(1):227-37
pubmed: 25971439
Front Cell Dev Biol. 2020 Nov 30;8:605996
pubmed: 33330507
J Bone Miner Res. 2011 Jul;26(7):1381-8
pubmed: 21538511
J Clin Endocrinol Metab. 2015 Oct;100(10):3625-32
pubmed: 26176801
PLoS Biol. 2016 Apr 01;14(4):e1002427
pubmed: 27035636

Auteurs

Margaux Gadion (M)

Université Paris Cité Faculté de Santé (Unité de formation et de recherche Odontologie et Unité de formation et de recherche de médecine) Paris France.
Assistance publique des hopitaux de Paris, Reference Center for Rare Diseases of the Calcium and Phosphate Metabolism, OSCAR Network, Fédération hospitalo-universitaire DDS-net, European reference network BOND, Department of Dental Medicine Bretonneau Hospital Paris France.

Agathe Hervé (A)

Université Paris Cité Faculté de Santé (Unité de formation et de recherche Odontologie et Unité de formation et de recherche de médecine) Paris France.
Assistance publique des hopitaux de Paris, Reference Center for Rare Diseases of the Calcium and Phosphate Metabolism, OSCAR Network, Fédération hospitalo-universitaire DDS-net, European reference network BOND, Department of Dental Medicine Bretonneau Hospital Paris France.

Julia Herrou (J)

Université Paris Cité Faculté de Santé (Unité de formation et de recherche Odontologie et Unité de formation et de recherche de médecine) Paris France.
Assistance publique des hopitaux de Paris, Reference Center for Rare Diseases of the Calcium and Phosphate Metabolism, OSCAR Network, European reference network BOND, Department of Rheumatology Cochin Hospital Paris France.

Anya Rothenbuhler (A)

Assistance publique des hopitaux de Paris, Reference Center for Rare Diseases of the Calcium and Phosphate Metabolism, OSCAR Network, Endo-European reference network and European reference network BOND, Department of Endocrinology and Diabetology for Children Bicêtre Paris Saclay Hospital Le Kremlin Bicêtre France.

Violaine Smail-Faugeron (V)

Université Paris Cité Faculté de Santé (Unité de formation et de recherche Odontologie et Unité de formation et de recherche de médecine) Paris France.
Assistance publique des hopitaux de Paris, Reference Center for Rare Diseases of the Calcium and Phosphate Metabolism, OSCAR Network, Fédération hospitalo-universitaire DDS-net, European reference network BOND, Department of Dental Medicine Bretonneau Hospital Paris France.

Frédéric Courson (F)

Université Paris Cité Faculté de Santé (Unité de formation et de recherche Odontologie et Unité de formation et de recherche de médecine) Paris France.
Assistance publique des hopitaux de Paris, Reference Center for Rare Diseases of the Calcium and Phosphate Metabolism, OSCAR Network, Fédération hospitalo-universitaire DDS-net, European reference network BOND, Department of Dental Medicine Bretonneau Hospital Paris France.

Agnès Linglart (A)

Assistance publique des hopitaux de Paris, Reference Center for Rare Diseases of the Calcium and Phosphate Metabolism, OSCAR Network, Endo-European reference network and European reference network BOND, Department of Endocrinology and Diabetology for Children Bicêtre Paris Saclay Hospital Le Kremlin Bicêtre France.
Université Paris Saclay Institut national de la santé et de la recherche médicale unité mixte de recherche 1185 Physiologie et physiopathologie endocrinienne Le Kremlin Bicêtre France.

Catherine Chaussain (C)

Université Paris Cité Faculté de Santé (Unité de formation et de recherche Odontologie et Unité de formation et de recherche de médecine) Paris France.
Assistance publique des hopitaux de Paris, Reference Center for Rare Diseases of the Calcium and Phosphate Metabolism, OSCAR Network, Fédération hospitalo-universitaire DDS-net, European reference network BOND, Department of Dental Medicine Bretonneau Hospital Paris France.
Université Paris Cité Laboratory Unité de recherche2496 Orofacial Pathologies, Imaging and Biotherapies Paris France.

Martin Biosse Duplan (M)

Université Paris Cité Faculté de Santé (Unité de formation et de recherche Odontologie et Unité de formation et de recherche de médecine) Paris France.
Assistance publique des hopitaux de Paris, Reference Center for Rare Diseases of the Calcium and Phosphate Metabolism, OSCAR Network, Fédération hospitalo-universitaire DDS-net, European reference network BOND, Department of Dental Medicine Bretonneau Hospital Paris France.
Institut Imagine, INSERM U1163 Paris France.

Classifications MeSH