{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,24]],"date-time":"2026-08-24T14:33:57Z","timestamp":1787582037223,"version":"build-2736575974"},"reference-count":14,"publisher":"The Endocrine Society","issue":"11","license":[{"start":{"date-parts":[[2023,4,21]],"date-time":"2023-04-21T00:00:00Z","timestamp":1682035200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/100013220","name":"Ultragenyx Pharmaceutical Inc","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100013220","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2023,10,18]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Context<\/jats:title>\n                  <jats:p>In an open-label, randomized, controlled, phase 3 trial in 61 children aged 1 to 12 years with X-linked hypophosphatemia (XLH), burosumab improved rickets vs continuing conventional therapy with active vitamin D and phosphate.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Objective<\/jats:title>\n                  <jats:p>We conducted an analysis to determine whether skeletal responses differed when switching to burosumab vs continuing higher or lower doses of conventional therapy.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>Conventional therapy dose groups were defined as higher-dose phosphate [greater than 40 mg\/kg] (HPi), lower-dose phosphate [40 mg\/kg or less] (LPi), higher-dose alfacalcidol [greater than 60 ng\/kg] or calcitriol [greater than 30 ng\/kg] (HD), and lower-dose alfacalcidol [60 ng\/kg or less] or calcitriol [30 ng\/kg or less] (LD).<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>At week 64, the Radiographic Global Impression of Change (RGI-C) for rickets was higher (better) in children randomly assigned to burosumab vs conventional therapy for all prebaseline dose groups: HPi (+1.72 vs +0.67), LPi (+2.14 vs +1.08), HD (+1.90 vs +0.94), LD (+2.11 vs +1.06). At week 64, the RGI-C for rickets was also higher in children randomly assigned to burosumab (+2.06) vs conventional therapy for all on-study dose groups: HPi (+1.03), LPi (+1.05), HD (+1.45), LD (+0.72). Serum alkaline phosphatase (ALP) also decreased in the burosumab-treated patients more than in the conventional therapy group, regardless of on-study phosphate and active vitamin D doses.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusion<\/jats:title>\n                  <jats:p>Prior phosphate or active vitamin D doses did not influence treatment response after switching to burosumab among children with XLH and active radiographic rickets. Switching from conventional therapy to burosumab improved rickets and serum ALP more than continuing either higher or lower doses of phosphate or active vitamin D.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1210\/clinem\/dgad230","type":"journal-article","created":{"date-parts":[[2023,4,21]],"date-time":"2023-04-21T21:07:47Z","timestamp":1682111267000},"page":"2990-2998","source":"Crossref","is-referenced-by-count":16,"title":["Burosumab vs Phosphate\/Active Vitamin D in Pediatric X-Linked Hypophosphatemia: A Subgroup Analysis by Dose Level"],"prefix":"10.1210","volume":"108","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7284-3467","authenticated-orcid":false,"given":"Erik A","family":"Imel","sequence":"first","affiliation":[{"name":"Departments of Medicine and Pediatrics, Indiana University School of Medicine , Indianapolis, IN 46202 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Francis H","family":"Glorieux","sequence":"additional","affiliation":[{"name":"Emeritus Director of Research, Shriners Hospitals for Children\u2013Canada , Montreal, QC H4A 0A9 , Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Michael P","family":"Whyte","sequence":"additional","affiliation":[{"name":"Center for Metabolic Bone Disease and Molecular Research, Shriners Hospitals for Children\u2013St. Louis , St Louis, MO 63110 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anthony A","family":"Portale","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, University of California, San Francisco , San Francisco, CA 94115 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Craig F","family":"Munns","sequence":"additional","affiliation":[{"name":"The University of Queensland, Queensland Children\u2019s Hospital , South Brisbane, QLD 4101 , Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ola","family":"Nilsson","sequence":"additional","affiliation":[{"name":"Division of Pediatric Endocrinology and Center for Molecular Medicine, Department of Women\u2019s and Children\u2019s Health, Karolinska Institutet and University Hospital , Stockholm 171 64 , Sweden"},{"name":"School of Medical Sciences and Department of Pediatrics, \u00d6rebro University and University Hospital , \u00d6rebro 701 85 , Sweden"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jill H","family":"Simmons","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Vanderbilt University Medical Center , Nashville, TN 37232 , USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Raja","family":"Padidela","sequence":"additional","affiliation":[{"name":"Paediatric Endocrinology, Royal Manchester Children\u2019s Hospital and Faculty of Biology, Medicine and Health, University of Manchester , Manchester M13 9PL , UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Noriyuki","family":"Namba","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Osaka Hospital, Japan Community Healthcare Organization , Osaka 553-0003 , Japan"},{"name":"Department of Pediatrics, Osaka University Graduate School of Medicine , Suita 565-0871 , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hae Il","family":"Cheong","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Hallym University Sacred Heart Hospital , Anyang-si, Gyeonggi-do , South Korea"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pisit","family":"Pitukcheewanont","sequence":"additional","affiliation":[{"name":"Children's Hospital Los Angeles, Los Angeles, CA and Keck School of Medicine of USC , Los Angeles, CA 90027 , 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