Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
. 2024 Oct;27(5):697-703.
doi: 10.1111/ocr.12787. Epub 2024 Apr 12.

Orthodontic treatment in children and adolescent patients with X-linked hypophosphatemia: A case-control study

Yann Janssens  1 , Martin Biosse Duplan  2   3 , Agnès Linglart  4   5 , Anya Rothenbuhler  4 , Catherine Chaussain  2   6 , Elvire Le Norcy  1   2   6
Affiliations

Orthodontic treatment in children and adolescent patients with X-linked hypophosphatemia: A case-control study

Yann Janssens et al. Orthod Craniofac Res. 2024 Oct.

Abstract

Objectives: X-linked hypophosphatemia (XLH) is a rare genetic disease that disturbs bone and teeth mineralization. It also affects craniofacial growth and patients with XLH often require orthodontic treatment. The aim of this study was to describe changes in the dental health of XLH children during orthodontic treatment compared with those in matched controls undergoing similar orthodontic procedures.

Materials and methods: For this retrospective case-control study, we included all individuals less than 16 years old diagnosed with XLH, orthodontically treated in our centre from 2016 to 2022 and pair-matched them to patients with no chronic or genetic conditions. Clinical and radiological parameters concerning their malocclusion, craniofacial discrepancy and the characteristics and iatrogenic effects of their orthodontic treatment were analysed.

Results: Fifteen XLH patients (mean age: 11.3 ± 2.1), pair-matched to 15 control patients were included. Orthodontic treatment was successfully conducted in XLH patients with slightly shorter duration and similar iatrogenic effects as in the control group, except for the occurrence of dental abscess during and after orthodontic tooth movement. XLH patients did not show more relapse than the controls.

Conclusion: Despite the presence of oral manifestations of XLH such as spontaneous abscesses, XLH patients can undergo orthodontic treatment with no obvious additional iatrogenic effects.

Keywords: XLH; case series; craniofacial growth; dental abscesses; dental anomalies; orthodontic treatment; rare skeletal disorder.

PubMed Disclaimer

References

REFERENCES

    1. Tenenhouse HS. X‐linked hypophosphataemia: a homologous disorder in humans and mice. Nephrol Dial Transplant. 1999;14:333‐341.
    1. Francis F, Hennig S, Korn B, et al. A gene (PEX) with homologies to endopeptidases is mutated in patients with X–linked hypophosphatemic rickets. Nat Genet. 1995;11:130‐136.
    1. Linglart A, Biosse‐Duplan M, Briot K, et al. Therapeutic management of hypophosphatemic rickets from infancy to adulthood. Endocr Connect. 2014;3:R13‐R30.
    1. Boukpessi T, Septier D, Bagga S, Garabedian M, Goldberg M, Chaussain‐Miller C. Dentin alteration of deciduous teeth in human hypophosphatemic rickets. Calcif Tissue Int. 2006;79:294‐300.
    1. Biosse Duplan M, Coyac BR, Bardet C, et al. Phosphate and vitamin D prevent periodontitis in X‐linked hypophosphatemia. J Dent Res. 2017;96:388‐395.

LinkOut - more resources