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REVIEW article

Front. Endocrinol.
Sec. Pediatric Endocrinology
Volume 15 - 2024 | doi: 10.3389/fendo.2024.1414509
This article is part of the Research Topic Insights in Pediatric Endocrinology: 2024 View all 6 articles

Meta-Analysis and Systematic Review: Burosumab as a Promising Treatment for Children with X-Linked Hypophosphatemia (XLH)

Provisionally accepted
Kangning Wang Kangning Wang Runze Zhang Runze Zhang Ziyi Chen Ziyi Chen Qing He Qing He *
  • School & Hospital of Stomatology, Wuhan University, Wuhan, China

The final, formatted version of the article will be published soon.

    The aim of this study was to evaluate the effectiveness of burosumab therapy in children with X-Linked Hypophosphatemia (XLH).We systematically reviewed literature from PubMed, Web of Science, The Cochrane Library, and Embase up until January 2024, using EndNote Web for study organization. The Newcastle-Ottawa scale guided quality assessment, while Revman software was used for data analysis and visualization. Study selection, 2 quality evaluation, and data aggregation were independently performed by three researchers.The meta-analysis encompassed ten studies, including eight cohort studies that examined burosumab's impact pre-and post-administration, and two randomized controlled trials comparing burosumab to standard therapy. The evidence from this review suggests burosumab's superiority in managing XLH in pediatric populations, particularly in improving key biochemical markers including 1,25-dihydroxyvitamin D (1,25-(OH)2D), phosphorus, and alkaline phosphatase (ALP), alongside improvements in the renal tubular maximum reabsorption rate of phosphate to glomerular filtration rate (TmP/GFR), and significant skeletal improvements as indicated by the rickets severity score (RSS) and the 6-minute walk test (6MWT).However, the long-term safety and effects, including height and quality of life (QOL) data, remains to be elucidated.: Burosumab has shown significant therapeutic effectiveness in treating children with XLH, highlighting its potential as a key treatment option. or obviously representative series of cases ✯, (b) potential for selection biases or not stated. g (a) community controls ✯,(b) hospital controls, (c) no description. h (a) no history of disease (endpoint) ✯, (b) no description of source. i (a) the exposures of interest (burosumab and active control) were adjusted for one confounder ( sex or age) ✯, (b) the exposures of interest (burosumab and active controll) were adjusted for two or more confounders ( sex or age and treatment compliance or severity of illness) ✯✯, c) no description. j (a) secure record (e.g., surgical records) ✯, (b)structured interview where blind to case/control status ✯, (c) interview not blinded to case/control status, (d) written self-report or medical record only, (e) no description. k (a) yes ✯, (b) no. l (a) same rate for both groups ✯, (b) non-respondents described, (c) rate different and no designation.

    Keywords: X-linked hypophosphatemia, Burosumab, pediatric, Meta-analysis, therapeutic effectiveness

    Received: 09 Apr 2024; Accepted: 08 Jul 2024.

    Copyright: © 2024 Wang, Zhang, Chen and He. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) or licensor are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

    * Correspondence: Qing He, School & Hospital of Stomatology, Wuhan University, Wuhan, China

    Disclaimer: All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article or claim that may be made by its manufacturer is not guaranteed or endorsed by the publisher.