{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,5]],"date-time":"2026-09-05T09:48:12Z","timestamp":1788601692255,"version":"build-2803163510"},"reference-count":73,"publisher":"The Endocrine Society","issue":"2","license":[{"start":{"date-parts":[[2024,7,25]],"date-time":"2024-07-25T00:00:00Z","timestamp":1721865600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"funder":[{"DOI":"10.13039\/100007560","name":"Chiesi","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100007560","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Cystinose-Stiftung"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,1,21]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Context<\/jats:title>\n                  <jats:p>The pathophysiology of cystinosis-associated metabolic bone disease is complex.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Objective<\/jats:title>\n                  <jats:p>We hypothesized a disturbed interaction between osteoblasts and osteoclasts.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>This binational cross-sectional multicenter study included 103 patients with cystinosis (61% children) with chronic kidney disease (CKD) stages 1 to 5D\/T at hospital clinics. Ten key bone markers were evaluated.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>Skeletal complications occurred in two-thirds of the patients, with adults having a 5-fold increased risk compared with children. Patients with CKD stages 1 to 3 showed reduced z-scores for serum phosphate and calcium and suppressed fibroblast growth factor 23 (FGF23) and parathyroid hormone levels, in conjunction with elevated bone-specific alkaline phosphatase levels. Serum phosphate was associated with estimated glomerular filtration rate, combined phosphate and active vitamin D treatment, and native vitamin D supplementation, while serum calcium was associated with age and dosage of active vitamin D. Sclerostin was generally elevated in children, and associated with age, FGF23 levels, and treatment with active vitamin D and growth hormone. The osteoclast marker tartrate-resistant acid phosphatase 5b was increased, and associated with age and treatment with active vitamin D. The ratio of soluble ligand of receptor activator of nuclear factor-\u03baB (sRANKL) and osteoprotegerin (OPG), a surrogate for the regulation of osteoclastogenesis by osteoblasts, was decreased and associated with phosphate and 1,25(OH)2D3 levels. These changes were only partly corrected after transplantation.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusion<\/jats:title>\n                  <jats:p>Bone health in cystinosis deteriorates with age, which is associated with increased osteoclast activity despite counter-regulation of osteoblasts via OPG\/RANKL, which in conjunction with elevated sclerostin levels and persistent rickets\/osteomalacia, may promote progressive bone loss.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1210\/clinem\/dgae502","type":"journal-article","created":{"date-parts":[[2024,7,25]],"date-time":"2024-07-25T10:59:11Z","timestamp":1721905151000},"page":"e218-e230","source":"Crossref","is-referenced-by-count":8,"title":["Cystinosis-Associated Metabolic Bone Disease Across Ages and CKD Stages 1 to 5D\/T"],"prefix":"10.1210","volume":"110","author":[{"given":"Johannes","family":"Lahring","sequence":"first","affiliation":[{"name":"Department of Pediatric Kidney, Liver and Metabolic Diseases, Pediatric Research Center, Hannover Medical School , D-30625 Hanover ,","place":["Germany"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-9203-7622","authenticated-orcid":false,"given":"Maren","family":"Leifheit-Nestler","sequence":"additional","affiliation":[{"name":"Department of Pediatric Kidney, Liver and Metabolic Diseases, Pediatric Research Center, Hannover Medical School , D-30625 Hanover ,","place":["Germany"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Annika","family":"Ewert","sequence":"additional","affiliation":[{"name":"Department of Pediatric Kidney, Liver and Metabolic Diseases, Pediatric Research Center, Hannover Medical School , D-30625 Hanover ,","place":["Germany"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nadine","family":"Herzig","sequence":"additional","affiliation":[{"name":"Department of Pediatric Orthopedics, Schoen Clinic Mu\u0308nchen Harlaching , D-81547 Munich ,","place":["Germany"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christian","family":"K\u00f6ppl","sequence":"additional","affiliation":[{"name":"Socialpediatric Center, Clinic Traunstein, Kliniken Su\u0308dostbayern AG , D-83278 Traunstein ,","place":["Germany"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Veronika","family":"Pott","sequence":"additional","affiliation":[{"name":"Department of Pediatric Kidney, Liver and Metabolic Diseases, Pediatric Research Center, Hannover Medical School , D-30625 Hanover ,","place":["Germany"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jun","family":"Oh","sequence":"additional","affiliation":[{"name":"Division of Pediatric Nephrology, University Children's Hospital Hamburg , D-20251 Hamburg ,","place":["Germany"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anja","family":"B\u00fcscher","sequence":"additional","affiliation":[{"name":"Department of Pediatrics II, University Hospital Essen , D-45152 Essen ,","place":["Germany"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Julia","family":"Thumfart","sequence":"additional","affiliation":[{"name":"Department of Pediatric Gastroenterology, Nephrology and Metabolic Diseases, Charit\u00e9 Universit\u00e4tsmedizin Berlin , D-13353 Berlin ,","place":["Germany"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lutz T","family":"Weber","sequence":"additional","affiliation":[{"name":"Pediatric Nephrology, Children's and Adolescents\u2019 Hospital, University of Cologne, Faculty of Medicine and University Hospital , D-50937 Cologne ,","place":["Germany"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Klaus","family":"Arbeiter","sequence":"additional","affiliation":[{"name":"Division of Pediatric Nephrology and Gastroenterology, Medical University Vienna , 1090 Viena ,","place":["Austria"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Birgit","family":"Acham-Roschitz","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Medical University Graz , 8036 Graz ,","place":["Austria"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Burkhard","family":"T\u00f6nshoff","sequence":"additional","affiliation":[{"name":"Department of Pediatrics I, University Children's Hospital Heidelberg , D-69120 Heidelberg ,","place":["Germany"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Miroslav","family":"Zivicnjak","sequence":"additional","affiliation":[{"name":"Department of Pediatric Kidney, Liver and Metabolic Diseases, Pediatric Research 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