{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,17]],"date-time":"2026-07-17T06:08:19Z","timestamp":1784268499370,"version":"3.55.0"},"reference-count":37,"publisher":"Oxford University Press (OUP)","issue":"7","license":[{"start":{"date-parts":[[2026,5,28]],"date-time":"2026-05-28T00:00:00Z","timestamp":1779926400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"funder":[{"name":"Kyowa Kirin Co., Ltd., Tokyo, Japan"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,6,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Burosumab has shown benefits in treating X-linked hypophosphatemia (XLH), a rare genetic bone disorder that causes rickets among pediatric patients. However, data in Chinese children with XLH are lacking. Therefore, we evaluated the effectiveness, safety, and pharmacokinetics of burosumab among Chinese children (aged 1-12\u00a0yr) with XLH in this open-label, multi-center, single-arm, phase 4 study. Of the 28 patients enrolled (12 male, 16 female), 27 completed the study. All patients received s.c. injections of burosumab at a starting dose of 0.8\u00a0mg\/kg every 2\u00a0wk for 64\u00a0wk. The mean\u2009\u00b1\u2009SD patient age was 5.68\u2009\u00b1\u20093.07\u00a0yr. Burosumab treatment increased the serum phosphorus level by 0.96\u2009\u00b1\u20090.27\u00a0mg\/dL from baseline during the burosumab dose cycles (average of week 2, 4, 8, 12, 16, 24, 32, 40, 52, and 64). The improvement from baseline (2.33\u2009\u00b1\u20090.24\u00a0mg\/dL) to week 64 (3.29\u2009\u00b1\u20090.30\u00a0mg\/dL) was significant (p\u2009&amp;lt;\u2009.05). The Radiograph Global Impression of Change was significantly improved at week 64 (1.90\u2009\u00b1\u20090.48; p\u2009&amp;lt;\u2009.0001 vs baseline), and the percentage predicted 6-min walking test among patients aged \u22655\u00a0yr with post-baseline values (n\u2009=\u200916) was significantly increased from 67.32%\u2009\u00b1\u20098.27% at baseline to 71.91%\u2009\u00b1\u20098.25% at week 64 (p\u2009&amp;lt;\u2009.05). All patients experienced treatment-emergent adverse events; most were mild to moderate. In conclusion, burosumab treatment in Chinese children with XLH corrected serum phosphorus levels and improved clinical parameters with tolerable safety profiles.<\/jats:p>","DOI":"10.1093\/jbmrpl\/ziag094","type":"journal-article","created":{"date-parts":[[2026,5,26]],"date-time":"2026-05-26T11:18:53Z","timestamp":1779794333000},"source":"Crossref","is-referenced-by-count":0,"title":["Effectiveness, safety, and pharmacokinetics of burosumab in Chinese children with X-linked hypophosphatemia: an open-label, single-arm phase 4 study"],"prefix":"10.1093","volume":"10","author":[{"given":"Wei","family":"Wu","sequence":"first","affiliation":[{"name":"Department of Pediatrics, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology , Wuhan 430030 ,","place":["China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4434-3462","authenticated-orcid":false,"given":"Yan","family":"Jiang","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Key Laboratory of Endocrinology, National Commission of Health, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences , Beijing 100730 ,","place":["China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhenlin","family":"Zhang","sequence":"additional","affiliation":[{"name":"Department of Osteoporosis and Bone Diseases, Shanghai Sixth People's Hospital Affiliated to Shanghai Jiao Tong University School of Medicine , Shanghai 200233 ,","place":["China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Li","family":"Liu","sequence":"additional","affiliation":[{"name":"Guangzhou Women and Children's Medical Center , Guangzhou 510623 ,","place":["China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wenjuan","family":"Qiu","sequence":"additional","affiliation":[{"name":"Department of Pediatric Endocrinology and Genetics, Xinhua Hospital, Shanghai Institute for Pediatric Research, Shanghai Jiao Tong University School of Medicine , Shanghai 200092 ,","place":["China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ying","family":"Ji","sequence":"additional","affiliation":[{"name":"Development and Regulatory Division, Kyowa Kirin China Pharmaceutical Co., Ltd. , Beijing 100027 ,","place":["China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maiko","family":"Sugimoto","sequence":"additional","affiliation":[{"name":"Development Division, Kyowa Kirin Co., Ltd. , Tokyo 100-0004 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chinwei","family":"Yung","sequence":"additional","affiliation":[{"name":"Development Division, Kyowa Kirin Co., Ltd. , Tokyo 100-0004 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"ChenRui","family":"Qiu","sequence":"additional","affiliation":[{"name":"Medical Affairs Department, Kyowa Kirin China Pharmaceutical Co., Ltd. , Beijing 100027 ,","place":["China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masaaki","family":"Kuriki","sequence":"additional","affiliation":[{"name":"Development Division, Kyowa Kirin Co., Ltd. , Tokyo 100-0004 ,","place":["Japan"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3748-4817","authenticated-orcid":false,"given":"Xiaoping","family":"Luo","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Tongji Hospital, Tongji Medical College, Huazhong University of Science and Technology , Wuhan 430030 ,","place":["China"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7768-5536","authenticated-orcid":false,"given":"Weibo","family":"Xia","sequence":"additional","affiliation":[{"name":"Department of Endocrinology, Key Laboratory of Endocrinology, National Commission of Health, State Key Laboratory of Complex Severe and Rare Diseases, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences , Beijing 100730 ,","place":["China"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2026,5,28]]},"reference":[{"issue":"7","key":"2026071701414699300_ref1","doi-asserted-by":"publisher","first-page":"1321","DOI":"10.1210\/js.2018-00365","article-title":"The lifelong impact of X-linked hypophosphatemia: results from a burden of disease survey","volume":"3","author":"Skrinar","year":"2019","journal-title":"J Endocr 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