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The present work describes the pharmacokinetics (PK) of burosumab and the pharmacokinetic\u2010pharmacodynamic (PK\u2010PD) relationship between burosumab and serum phosphorus in adult and pediatric patients with XLH. A total of 2844 measurable serum concentrations of burosumab and 6047 measurable serum concentrations of phosphorus in 277 subjects from 9 clinical studies were included in the population PK and PK\u2010PD modeling. The serum concentration of burosumab following a subcutaneous administration was well described by a population PK model comprising a first\u2010order absorption, 1\u2010compartmental distribution, and a linear elimination. The relationship between serum burosumab and serum phosphorus was adequately described by a sigmoid maximal efficacy model. Body weight was the only covariate associated with PK and PK\u2010PD parameters. No other intrinsic factors affected PK or PK\u2010PD relationship in adult and pediatric patients with XLH. Further simulations helped to guide the dosing regimen of burosumab in adult and pediatric patients with XLH including age groups with no clinical data.<\/jats:p>","DOI":"10.1002\/jcph.1950","type":"journal-article","created":{"date-parts":[[2021,8,5]],"date-time":"2021-08-05T17:38:44Z","timestamp":1628185124000},"page":"87-98","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":14,"title":["Population Pharmacokinetics and Pharmacodynamics of Burosumab in Adult and Pediatric Patients With X\u2010linked Hypophosphatemia"],"prefix":"10.1002","volume":"62","author":[{"given":"Sun Ku","family":"Lee","sequence":"first","affiliation":[{"name":"Ultragenyx Pharmaceutical Inc.  Novato California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nathalie H.","family":"Gosselin","sequence":"additional","affiliation":[{"name":"Certara Strategic Consulting  Princeton New Jersey USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Julie","family":"Taylor","sequence":"additional","affiliation":[{"name":"Ultragenyx Pharmaceutical Inc.  Novato California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mary Scott","family":"Roberts","sequence":"additional","affiliation":[{"name":"Ultragenyx Pharmaceutical Inc.  Novato California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kathleen","family":"McKeever","sequence":"additional","affiliation":[{"name":"Ultragenyx Pharmaceutical Inc.  Novato California USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jack","family":"Shi","sequence":"additional","affiliation":[{"name":"Ultragenyx Pharmaceutical Inc.  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