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Objectives were to characterize effects of estimated glomerular filtration rate (eGFR) on oral clearance (CL), predict doses in mild to moderate renal impairment and in pediatric patients with FD, and to improve designs of FD studies. A 2\u2010compartment model was fit to data from 260 subjects with\/without FD and iteratively refined with evolving data. FD, eGFR, and weight affected CL, while weight and FD affected volume. Optimal sampling theory was used to choose pharmacokinetic sampling times for pediatric studies. Doses in patients with renal impairment and in pediatrics were determined by targeting exposure in adults receiving migalastat 123\u00a0mg every other day. A clinical study was conducted in 20 adolescent patients with FD \u226545\u00a0kg. eGFR had the largest effect on CL. Simulations showed that exposures in moderate renal impairment were within phase 2\u20133 exposures; patients aged 2\u201317 years require weight\u2010based dosing; and predicted exposures in adolescent patients \u226545\u00a0kg receiving migalastat 123\u00a0mg every other day were similar to adults (data confirmed in a clinical study). Model\u2010informed drug development optimized dosing and design of clinical studies and supported that no dose adjustments were needed in patients with mild to moderate renal impairment or in adolescent patients \u226545\u00a0kg.<\/jats:p>","DOI":"10.1002\/cpdd.1160","type":"journal-article","created":{"date-parts":[[2022,11,4]],"date-time":"2022-11-04T15:59:28Z","timestamp":1667577568000},"page":"1367-1381","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Population Pharmacokinetics of Oral Migalastat in Adolescents and Adults With and Without Renal Impairment"],"prefix":"10.1002","volume":"11","author":[{"given":"Cathrine","family":"Leonowens","sequence":"first","affiliation":[{"name":"Nuventra LLC  Durham North Carolina USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Virginia","family":"Schmith","sequence":"additional","affiliation":[{"name":"Nuventra LLC  Durham North Carolina USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jie","family":"Zhou","sequence":"additional","affiliation":[{"name":"Nuventra LLC  Exton Pennsylvania USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yi Shuan","family":"Wu","sequence":"additional","affiliation":[{"name":"Nuventra LLC  Durham North Carolina USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Vijay","family":"Ivaturi","sequence":"additional","affiliation":[{"name":"Pumas\u2010AI, Inc.  Baltimore Maryland USA"},{"name":"University of Maryland  Baltimore Maryland USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Franklin K.","family":"Johnson","sequence":"additional","affiliation":[{"name":"Amicus Therapeutics, Inc.  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