Improved Oral Health in Adults with X-Linked Hypophosphatemia Treated with Burosumab.


Journal

The Journal of clinical endocrinology and metabolism
ISSN: 1945-7197
Titre abrégé: J Clin Endocrinol Metab
Pays: United States
ID NLM: 0375362

Informations de publication

Date de publication:
12 Jun 2024
Historique:
received: 27 02 2024
revised: 03 06 2024
accepted: 07 06 2024
medline: 12 6 2024
pubmed: 12 6 2024
entrez: 12 6 2024
Statut: aheadofprint

Résumé

X-linked hypophosphatemia (XLH) is a rare genetic bone disease affecting both children and adults, with oral manifestations such as spontaneous dental infections. The main treatments for XLH are conventional treatment (CT) with oral phosphate salts and active vitamin D supplementation, and burosumab, an antibody targeting Fibroblast Growth Factor 23 (FGF23). While the beneficial effect of CT on oral manifestations is established, the effect of burosumab on oral health is unknown, especially in adults. We aimed to compare the oral health (number of missing or endodontically treated teeth and presence of periodontal disease) and incidence of endodontic infections of adult patients with XLH according to their treatment's modalities (no treatment, CT, or burosumab). This was achieved through a single-center, retrospective analysis of oral health data from 44 patients who had undergone dental monitoring for at least 6 months. Oral health varied according to the proportion of their adult life spent under treatment for XLH and the incidence of dental infections during follow-up was influenced by the type of treatment received. There was a 55.9% reduction of infections during CT and an 86.4% reduction during burosumab treatment compared to periods with no treatment (P < 0.0001). Comparing treatment and non-treatment periods within the same patient showed a strong association between burosumab treatment and decreased infection incidence (0.006 vs 0.09 infection per month, P<0.01). We observed that adults with XLH treated with burosumab developed fewer endodontic infections during dental follow-up than patients who were untreated or received CT.

Identifiants

pubmed: 38864457
pii: 7691585
doi: 10.1210/clinem/dgae398
pii:
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Informations de copyright

© The Author(s) 2024. Published by Oxford University Press on behalf of the Endocrine Society. All rights reserved. For commercial re-use, please contact reprints@oup.com for reprints and translation rights for reprints. All other permissions can be obtained through our RightsLink service via the Permissions link on the article page on our site—for further information please contact journals.permissions@oup.com. See the journal About page for additional terms.

Auteurs

Agathe Hervé (A)

UFR Odontologie, Université Paris Cité & AP-HP, Reference center for rare diseases of the calcium and phosphate metabolism, OSCAR network, FHU DDS-net, ERN BOND, Department of dental medicine, Bretonneau hospital, Paris, France.

Margaux Gadion (M)

UFR Odontologie, Université Paris Cité & AP-HP, Reference center for rare diseases of the calcium and phosphate metabolism, OSCAR network, FHU DDS-net, ERN BOND, Department of dental medicine, Bretonneau hospital, Paris, France.

Julia Herrou (J)

UFR de médecine, Université Paris Cité & AP-HP, Reference center for rare diseases of the calcium and phosphate metabolism, OSCAR network, ERN BOND, Department of rheumatology, Cochin hospital, Paris, France.

Mathieu Izart (M)

UFR Odontologie, Université Paris Cité & AP-HP, Reference center for rare diseases of the calcium and phosphate metabolism, OSCAR network, FHU DDS-net, ERN BOND, Department of dental medicine, Bretonneau hospital, Paris, France.

Agnès Linglart (A)

AP-HP, Reference center for rare diseases of the calcium and phosphate metabolism, OSCAR network, Endo-ERN and ERN BOND, Department of endocrinology and diabetology for children, Bicêtre Paris Saclay hospital & Université Paris Saclay, INSERM UMR 1185, Le Kremlin Bicêtre, France.

Martine Cohen-Solal (M)

AP-HP, Reference center for rare bone diseases, OSCAR network, Department of Rheumatology, Lariboisiere hospital & Université Paris Cité, INSERM 1132, Paris, France.

Anne-Lise Lecoq (AL)

AP-HP, Reference center for rare diseases of the calcium and phosphate metabolism, OSCAR network, Endo-ERN and ERN BOND, Department of endocrinology, Bicêtre Paris Saclay hospital, & Université Paris Saclay, INSERM UMR 1185, Le Kremlin Bicêtre, France.

Peter Kamenicky (P)

AP-HP, Reference center for rare diseases of the calcium and phosphate metabolism, OSCAR network, Endo-ERN and ERN BOND, Department of endocrinology, Bicêtre Paris Saclay hospital, & Université Paris Saclay, INSERM UMR 1185, Le Kremlin Bicêtre, France.

Karine Briot (K)

UFR de médecine, Université Paris Cité & AP-HP, Reference center for rare diseases of the calcium and phosphate metabolism, OSCAR network, ERN BOND, Department of rheumatology, Cochin hospital, Paris, France.

Catherine Chaussain (C)

UFR Odontologie, Université Paris Cité & AP-HP, Reference center for rare diseases of the calcium and phosphate metabolism, OSCAR network, FHU DDS-net, ERN BOND, Department of dental medicine, Bretonneau hospital, Paris, France.
Université Paris Cité, Laboratory URP2496 Orofacial Pathologies, Imaging and Biotherapies, Paris, France.

Martin Biosse Duplan (M)

UFR Odontologie, Université Paris Cité & AP-HP, Reference center for rare diseases of the calcium and phosphate metabolism, OSCAR network, FHU DDS-net, ERN BOND, Department of dental medicine, Bretonneau hospital, Paris, France.
Institut Imagine, INSERM 1163, Paris, France.

Classifications MeSH