Improved Oral Health in Adults With X-Linked Hypophosphatemia Treated With Burosumab

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Abstract
Context: X-linked hypophosphatemia (XLH) is a rare genetic bone disease affecting both children and adults, with oral manifestations such as spontaneous dental infections. The main treatments for XLH are conventional treatment (CT) with oral phosphate salts and active vitamin D supplementation and burosumab, an antibody targeting fibroblast growth factor 23. While the beneficial effect of CT on oral manifestations is established, the effect of burosumab on oral health is unknown, especially in adults. Objective: We aimed to compare the oral health (number of missing or endodontically treated teeth and presence of periodontal disease) and incidence of endodontic infections of adult patients with XLH according to their treatment's modalities (no treatment, CT, or burosumab). Methods: This was achieved through a single-center, retrospective analysis of oral health data from 44 patients who had undergone dental monitoring for at least 6 months. Results: Oral health varied according to the proportion of their adult life spent under treatment for XLH, and the incidence of dental infections during follow-up was influenced by the type of treatment received. There was a 55.9% reduction of infections during CT and an 86.4% reduction during burosumab treatment compared to periods with no treatment (P < .0001). Comparing treatment and nontreatment periods within the same patient showed a strong association between burosumab treatment and decreased infection incidence (.006 vs .09 infection per month, P < .01). Conclusion: We observed that adults with XLH treated with burosumab developed fewer endodontic infections during dental follow-up than patients who were untreated or received CT.

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