Dental health of pediatric patients with X-linked hypophosphatemia (XLH) after three years of burosumab therapy

Autor: Rafi Brener, Leonid Zeitlin, Yael Lebenthal, Avivit Brener
Jazyk: angličtina
Rok vydání: 2022
Předmět:
Zdroj: Frontiers in Endocrinology, Vol 13 (2022)
Druh dokumentu: article
ISSN: 1664-2392
DOI: 10.3389/fendo.2022.947814
Popis: An inactivating PHEX gene mutation with the resultant accumulation of several mineralization-inhibiting proteins (e.g., FGF23) causes skeletal and dental morbidity in X-linked hypophosphatemia (XLH). This prospective case-control study explored the effect of burosumab, an anti-FGF23 antibody, on dental health of children with XLH. Ten children (age 4.3-15 years) with XLH underwent burosumab treatment per protocol. Assessment of their dental status at treatment initiation and after 1 and 3 years of treatment included clinical, laboratory and radiographic evaluation of rickets and dentition. Orthopantomographic examinations of ten healthy sex- and age-matched controls were selected for comparison. Coronal and pulp dimensions of a selected permanent mandibular molar were measured with Planmeca Romexis® software. One year of treatment led to improvement of height z-score (p=0.019) and healing of the rickets (p
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