Orthodontic treatment of a nine-year-old patient with hypophosphatemic rickets diagnosed since the age of two: A case report

Makrygiannakis, Miltiadis A; Dastoori, Mahmoud; Athanasiou, Athanasios E.

doi:10.1016/j.ortho.2020.06.003

Cited by 4 publications

(2 citation statements)

References 15 publications

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“…No dental or orthodontic complications were detected during or after treatment. A recent case report also reports no orthodontic complications during treatment (Makrygiannakis et al, 2020). There is little published on the implications of orthodontic treatment in patients with XLH.…”

Section: Clinical Featuresmentioning

confidence: 99%

X-linked hypophosphatemic rickets: Orthodontic considerations and management. A case report

2021

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X-linked hypophosphatemic rickets (XLH) is a rare condition affecting bone metabolism. It has characteristic dental features such as poorly mineralised dentine, spontaneous abscess formation in the absence of caries and taurodontism. There are limited published data about patients with this condition undergoing orthodontic treatment, and there is no clear guideline on the suitability of orthodontic treatment in this cohort. We present a case report of a patient with XLH with a confirmed PHEX gene mutation undergoing orthodontic treatment and clinical recommendations to support treatment.

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Section: Clinical Featuresmentioning

confidence: 99%

X-linked hypophosphatemic rickets: Orthodontic considerations and management. A case report

2021

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“…Certain dental malocclusions are more common, such as an open bite, maxillary retrognathism and impacted or ectopic maxillary canines [12][13][14]. Nevertheless, there are few reports of orthodontic treatment of patients with XLH [15]. Radiographic examinations of deciduous and permanent teeth reveal shorter roots, root dysplasia, enlarged pulp chambers and high pulp horns extending to the dentin-enamel junction (DEJ) [16].…”

Section: Introductionmentioning

confidence: 99%

Characteristics of oral health of patients with X-linked hypophosphatemia: case reports and literature review

Arhar,

Pavlič,

Hočevar

2024

BDJ Open

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Background Oral health is impaired in X-linked hypophosphatemia (XLH), resulting in delayed dental development, malocclusion, and radiographic abnormalities. This study investigates the oral manifestations in Slovenian XLH patients, focusing on enamel and dentin abnormalities and a literature review of spontaneous periapical abscesses in XLH cases. Objectives To report XLH patients with specific oral signs and symptoms, histological analysis of affected teeth, and review of reported cases of XLH patients with spontaneous periapical abscesses. Methods Case reports: Seven XLH patients from the National Registry of Patients with Rare Diseases underwent a detailed oral examination, including X-ray reviews. The patients who were expected to have tooth exfoliation or extraction were asked to donate their teeth for histological analysis by scanning electron microscopy. Literature search: A literature search of four electronic databases and a manual bibliography search aimed to identify documented cases of XLH with periapical abscesses up to January 21, 2024. Inclusion criteria were confirmed XLH patients with periapical abscesses in English peer-reviewed publications. Results Tooth samples from three XLH patients showed reduced dentin mineralisation, affecting one-third to one-half of the outer dentin. Inadequate mineralisation, uneven dentin tubules, and cracks and chipping in the enamel were observed, indicating mineralisation deviations. Similar cracks extended into the dentin and were also present in the root of the examined tooth. Based on the content of the 75 items identified in the search, spontaneous abscesses are not uncommon in patients with XLH. Conclusions XLH significantly affects patients’ lives and requires lifelong treatment. Dental examinations consistently revealed oral problems, including malocclusion. Histological analysis confirmed structural changes, especially in the dentin. Despite continued treatment, XLH patients may have an increased risk of oral pathologies. Further research is needed to understand the impact of XLH and its treatment on dental health.

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

Janssens,

Duplan,

Linglart

et al. 2024

Orthod Craniofacial Res

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ObjectivesX‐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 MethodsFor 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.ResultsFifteen 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.ConclusionDespite the presence of oral manifestations of XLH such as spontaneous abscesses, XLH patients can undergo orthodontic treatment with no obvious additional iatrogenic effects.

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Orthodontic treatment of a nine-year-old patient with hypophosphatemic rickets diagnosed since the age of two: A case report

Cited by 4 publications

References 15 publications

X-linked hypophosphatemic rickets: Orthodontic considerations and management. A case report

X-linked hypophosphatemic rickets: Orthodontic considerations and management. A case report

Characteristics of oral health of patients with X-linked hypophosphatemia: case reports and literature review

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

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