» Articles » PMID: 40003246

Dental Manifestations in Children Affected by Hypophosphatemic Rickets: A Systematic Review and Meta-Analysis

Overview
Specialty Health Services
Date 2025 Feb 26
PMID 40003246
Authors
Affiliations
Soon will be listed here.
Abstract

Background: Hypophosphatemic rickets (HR) is a bone disorder affecting phosphate-calcium metabolism, with both skeletal and dental manifestations. This review aims to analyze dental manifestations of HR in children and, where possible, compare them to those in healthy children or affected adults.

Methods: The protocol was registered at PROSPERO (CRD42024596022). The study conformed to the PRISMA guidelines. Three databases were searched for studies reporting the prevalence or incidence of any dental manifestation in children with HR. Risk of bias was assessed using JBI, RoB 2.0, and ROBINS-E tools, and Stata/SE 18.0 was used for meta-analysis. Meta-regression was used to examine the effects of therapy duration and mean age on dental manifestations' prevalence. The study received no funding.

Results: A total of 1308 records were identified, with 660 screened after removing duplicates. Forty-six studies were eligible for full-text evaluation; sixteen were included in the qualitative analysis and twelve in the meta-analysis. The dental manifestations observed included dental abscesses, developmental defects of enamel and dentin, dental caries, taurodontism, and large pulp chambers. Dental abscesses were the most common manifestation, with a pooled prevalence of 0.39. Meta-regression showed no association between therapy duration and abscess occurrence but revealed a negative association between mean age and abscess prevalence.

Conclusions: Dental abscesses were the most frequent manifestation in children with HR. The role of therapy in improving oral health remains unclear due to insufficient data, indicating a need for further studies on the impact of HR on children's oral health.

References
1.
Linglart A, Biosse-Duplan M, Briot K, Chaussain C, Esterle L, Guillaume-Czitrom S . Therapeutic management of hypophosphatemic rickets from infancy to adulthood. Endocr Connect. 2014; 3(1):R13-30. PMC: 3959730. DOI: 10.1530/EC-13-0103. View

2.
Souza M, Soares Junior L, Santos M, Vaisbich M . Dental abnormalities and oral health in patients with Hypophosphatemic rickets. Clinics (Sao Paulo). 2010; 65(10):1023-6. PMC: 2972601. DOI: 10.1590/s1807-59322010001000017. View

3.
Baroncelli G, Comberiati P, Aversa T, Baronio F, Cassio A, Chiarito M . Diagnosis, treatment, and management of rickets: a position statement from the Bone and Mineral Metabolism Group of the Italian Society of Pediatric Endocrinology and Diabetology. Front Endocrinol (Lausanne). 2024; 15:1383681. PMC: 11066174. DOI: 10.3389/fendo.2024.1383681. View

4.
Herve A, Gadion M, Herrou J, Izart M, Linglart A, Cohen-Solal M . Improved Oral Health in Adults with X-Linked Hypophosphatemia Treated with Burosumab. J Clin Endocrinol Metab. 2024; . DOI: 10.1210/clinem/dgae398. View

5.
Ariceta G, Beck-Nielsen S, Boot A, Brandi M, Briot K, Collantes C . The International X-Linked Hypophosphatemia (XLH) Registry: first interim analysis of baseline demographic, genetic and clinical data. Orphanet J Rare Dis. 2023; 18(1):304. PMC: 10523658. DOI: 10.1186/s13023-023-02882-4. View