Burosumab and Dental Abscesses in Children With X‐Linked Hypophosphatemia
ABSTRACT 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. Hypomineraliz...
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| Format: | Article |
| Language: | English |
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Wiley
2022-11-01
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| Series: | JBMR Plus |
| Subjects: | |
| Online Access: | https://doi.org/10.1002/jbm4.10672 |
| _version_ | 1798018827877875712 |
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| author | Margaux Gadion Agathe Hervé Julia Herrou Anya Rothenbuhler Violaine Smail‐Faugeron Frédéric Courson Agnès Linglart Catherine Chaussain Martin Biosse Duplan |
| author_facet | Margaux Gadion Agathe Hervé Julia Herrou Anya Rothenbuhler Violaine Smail‐Faugeron Frédéric Courson Agnès Linglart Catherine Chaussain Martin Biosse Duplan |
| author_sort | Margaux Gadion |
| collection | DOAJ |
| description | ABSTRACT 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; p = 0.04). Burosumab treatment appears to be associated with a reduction in the number of dental abscesses in XLH children, compared with CT. © 2022 The Authors. JBMR Plus published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research. |
| first_indexed | 2024-04-11T16:30:40Z |
| format | Article |
| id | doaj.art-ba5b9067e91d46b9823e2e97a6806941 |
| institution | Directory Open Access Journal |
| issn | 2473-4039 |
| language | English |
| last_indexed | 2024-04-11T16:30:40Z |
| publishDate | 2022-11-01 |
| publisher | Wiley |
| record_format | Article |
| series | JBMR Plus |
| spelling | doaj.art-ba5b9067e91d46b9823e2e97a68069412022-12-22T04:14:03ZengWileyJBMR Plus2473-40392022-11-01611n/an/a10.1002/jbm4.10672Burosumab and Dental Abscesses in Children With X‐Linked HypophosphatemiaMargaux Gadion0Agathe Hervé1Julia Herrou2Anya Rothenbuhler3Violaine Smail‐Faugeron4Frédéric Courson5Agnès Linglart6Catherine Chaussain7Martin Biosse Duplan8Université Paris Cité Faculté de Santé (Unité de formation et de recherche Odontologie et Unité de formation et de recherche de médecine) Paris FranceUniversité Paris Cité Faculté de Santé (Unité de formation et de recherche Odontologie et Unité de formation et de recherche de médecine) Paris FranceUniversité Paris Cité Faculté de Santé (Unité de formation et de recherche Odontologie et Unité de formation et de recherche de médecine) Paris FranceAssistance 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 FranceUniversité Paris Cité Faculté de Santé (Unité de formation et de recherche Odontologie et Unité de formation et de recherche de médecine) Paris FranceUniversité Paris Cité Faculté de Santé (Unité de formation et de recherche Odontologie et Unité de formation et de recherche de médecine) Paris FranceAssistance 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 FranceUniversité Paris Cité Faculté de Santé (Unité de formation et de recherche Odontologie et Unité de formation et de recherche de médecine) Paris FranceUniversité Paris Cité Faculté de Santé (Unité de formation et de recherche Odontologie et Unité de formation et de recherche de médecine) Paris FranceABSTRACT 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; p = 0.04). Burosumab treatment appears to be associated with a reduction in the number of dental abscesses in XLH children, compared with CT. © 2022 The Authors. JBMR Plus published by Wiley Periodicals LLC on behalf of American Society for Bone and Mineral Research.https://doi.org/10.1002/jbm4.10672DENTAL BIOLOGY|MATRIX MINERALIZATIONBONE MATRIXDISORDERS OF CALCIUM/PHOSPHATE METABOLISMPTH/VIT D/FGF23CELL/TISSUE SIGNALING – ENDOCRINE PATHWAYS |
| spellingShingle | Margaux Gadion Agathe Hervé Julia Herrou Anya Rothenbuhler Violaine Smail‐Faugeron Frédéric Courson Agnès Linglart Catherine Chaussain Martin Biosse Duplan Burosumab and Dental Abscesses in Children With X‐Linked Hypophosphatemia JBMR Plus DENTAL BIOLOGY |MATRIX MINERALIZATION BONE MATRIX DISORDERS OF CALCIUM/PHOSPHATE METABOLISM PTH/VIT D/FGF23 CELL/TISSUE SIGNALING – ENDOCRINE PATHWAYS |
| title | Burosumab and Dental Abscesses in Children With X‐Linked Hypophosphatemia |
| title_full | Burosumab and Dental Abscesses in Children With X‐Linked Hypophosphatemia |
| title_fullStr | Burosumab and Dental Abscesses in Children With X‐Linked Hypophosphatemia |
| title_full_unstemmed | Burosumab and Dental Abscesses in Children With X‐Linked Hypophosphatemia |
| title_short | Burosumab and Dental Abscesses in Children With X‐Linked Hypophosphatemia |
| title_sort | burosumab and dental abscesses in children with x linked hypophosphatemia |
| topic | DENTAL BIOLOGY |MATRIX MINERALIZATION BONE MATRIX DISORDERS OF CALCIUM/PHOSPHATE METABOLISM PTH/VIT D/FGF23 CELL/TISSUE SIGNALING – ENDOCRINE PATHWAYS |
| url | https://doi.org/10.1002/jbm4.10672 |
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