{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,28]],"date-time":"2026-09-28T20:13:43Z","timestamp":1790626423459,"version":"4.1.0"},"reference-count":57,"publisher":"The Endocrine Society","issue":"12","license":[{"start":{"date-parts":[[2022,9,16]],"date-time":"2022-09-16T00:00:00Z","timestamp":1663286400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"funder":[{"name":"Kyowa Kirin"},{"DOI":"10.13039\/501100006687","name":"Universit\u00e9 Claude Bernard Lyon 1","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100006687","id-type":"DOI","asserted-by":"crossref"}]},{"name":"Research Foundation"},{"name":"BoneTax","award":["ANR-20-CE14-00036-01"],"award-info":[{"award-number":["ANR-20-CE14-00036-01"]}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2022,11,25]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Context<\/jats:title>\n                  <jats:p>X-linked hypophosphatemia (XLH) is a rare genetic disease caused by a primary excess of fibroblast growth factor 23 (FGF23). FGF23 has been associated with inflammation and impaired osteoclastogenesis, but these pathways have not been investigated in XLH.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Objective<\/jats:title>\n                  <jats:p>This work aimed to evaluate whether XLH patients display peculiar inflammatory profile and increased osteoclastic activity.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>We performed a prospective, multicenter, cross-sectional study analyzing transcript expression of 8 inflammatory markers (Il6, Il8, Il1\u03b2, CXCL1, CCL2, CXCR3, Il1R, Il6R) by real-time quantitative polymerase chain reaction on peripheral blood mononuclear cells (PBMCs) purified from total blood samples extracted from patients and healthy control individuals. The effect of native\/active vitamin D on osteoclast formation was also assessed in vitro from XLH patients\u2019 PBMCs.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>In total, 28 XLH patients (17 children, among them 6 undergoing standard of care [SOC] and 11 burosumab therapy) and 19 controls were enrolled. Expression of most inflammatory markers was significantly increased in PBMCs from XLH patients compared to controls. No differences were observed between the burosumab and SOC subgroups. Osteoclast formation was significantly impaired in XLH patients. XLH mature osteoclasts displayed higher levels of inflammatory markers, being however lower in cells derived from the burosumab subgroup (as opposed to SOC).<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusion<\/jats:title>\n                  <jats:p>We describe for the first time a peculiar inflammatory profile in XLH. Since XLH patients have a propensity to develop arterial hypertension, obesity, and enthesopathies, and because inflammation can worsen these clinical outcomes, we hypothesize that inflammation may play a critical role in these extraskeletal complications of XLH.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1210\/clinem\/dgac543","type":"journal-article","created":{"date-parts":[[2022,9,16]],"date-time":"2022-09-16T15:57:55Z","timestamp":1663343875000},"page":"3275-3286","source":"Crossref","is-referenced-by-count":15,"title":["X-Linked Hypophosphatemia, Not Only a Skeletal Disease But Also a Chronic Inflammatory State"],"prefix":"10.1210","volume":"107","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-8418-1926","authenticated-orcid":false,"given":"Marie-No\u00eblle","family":"M\u00e9aux","sequence":"first","affiliation":[{"name":"Service de N\u00e9phrologie, Rhumatologie et Dermatologie P\u00e9diatriques, Centre de R\u00e9f\u00e9rence des Maladies Rares du Calcium et du Phosphore, Centre de R\u00e9f\u00e9rence des Maladies R\u00e9nales Rares Fili\u00e8res Maladies Rares ORKID, OSCAR et ERN ERK-Net, CHU de Lyon , 69 500 Bron , France"},{"name":"INSERM, UMR 1033, Facult\u00e9 de M\u00e9decine Lyon Est, Universit\u00e9 Claude Bernard Lyon 1 , 69 008 Lyon , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Candide","family":"Alioli","sequence":"additional","affiliation":[{"name":"INSERM, UMR 1033, Facult\u00e9 de M\u00e9decine Lyon Est, Universit\u00e9 Claude Bernard Lyon 1 , 69 008 Lyon , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Agn\u00e8s","family":"Linglart","sequence":"additional","affiliation":[{"name":"AP-HP, Universit\u00e9 Paris Saclay, INSERM, Service d\u2019endocrinologie et diab\u00e8te de l\u2019enfant, Centre de R\u00e9f\u00e9rence des Maladies Rares du Calcium et du Phosphore, Fili\u00e8re de Sant\u00e9 Maladies Rares OSCAR, ERN endoRARE et BOND, Plateforme d\u2019expertise des maladies rares Paris Saclay, H\u00f4pital Bic\u00eatre Paris-Saclay , 94 270 Le Kremlin-Bic\u00eatre , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sandrine","family":"Lemoine","sequence":"additional","affiliation":[{"name":"D\u00e9partement de N\u00e9phrologie, Service d\u2019exploration Fonctionnelle r\u00e9nale, H\u00f4pital Edouard Herriot , 69 003 Lyon , France"},{"name":"Facult\u00e9 de M\u00e9decine Lyon Est, Universit\u00e9 Claude Bernard Lyon 1 , 69 003 Lyon , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emmanuelle","family":"Vignot","sequence":"additional","affiliation":[{"name":"Facult\u00e9 de M\u00e9decine Lyon Est, Universit\u00e9 Claude Bernard Lyon 1 , 69 003 Lyon , France"},{"name":"Service de Rhumatologie, H\u00f4pital Edouard Herriot , 69 003 Lyon , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Aur\u00e9lia","family":"Bertholet-Thomas","sequence":"additional","affiliation":[{"name":"Service de N\u00e9phrologie, Rhumatologie et Dermatologie P\u00e9diatriques, Centre de R\u00e9f\u00e9rence des Maladies Rares du Calcium et du Phosphore, Centre de R\u00e9f\u00e9rence des Maladies R\u00e9nales Rares Fili\u00e8res Maladies Rares ORKID, OSCAR et ERN ERK-Net, CHU de Lyon , 69 500 Bron , France"},{"name":"INSERM, UMR 1033, Facult\u00e9 de M\u00e9decine Lyon Est, Universit\u00e9 Claude Bernard Lyon 1 , 69 008 Lyon , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Olivier","family":"Peyruchaud","sequence":"additional","affiliation":[{"name":"INSERM, UMR 1033, Facult\u00e9 de M\u00e9decine Lyon Est, Universit\u00e9 Claude Bernard Lyon 1 , 69 008 Lyon , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sacha","family":"Flammier","sequence":"additional","affiliation":[{"name":"Service de N\u00e9phrologie, Rhumatologie et Dermatologie P\u00e9diatriques, Centre de R\u00e9f\u00e9rence des Maladies Rares du Calcium et du Phosphore, Centre de R\u00e9f\u00e9rence des Maladies R\u00e9nales Rares Fili\u00e8res Maladies Rares ORKID, OSCAR et ERN ERK-Net, CHU de Lyon , 69 500 Bron , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Irma","family":"Machuca-Gayet","sequence":"additional","affiliation":[{"name":"INSERM, UMR 1033, Facult\u00e9 de M\u00e9decine Lyon Est, Universit\u00e9 Claude Bernard Lyon 1 , 69 008 Lyon , France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0578-2529","authenticated-orcid":false,"given":"Justine","family":"Bacchetta","sequence":"additional","affiliation":[{"name":"Service de N\u00e9phrologie, Rhumatologie et Dermatologie P\u00e9diatriques, Centre de R\u00e9f\u00e9rence des Maladies Rares du Calcium et du Phosphore, Centre de R\u00e9f\u00e9rence des Maladies R\u00e9nales Rares Fili\u00e8res Maladies Rares ORKID, OSCAR et ERN ERK-Net, CHU de Lyon , 69 500 Bron , France"},{"name":"INSERM, UMR 1033, Facult\u00e9 de M\u00e9decine Lyon Est, Universit\u00e9 Claude Bernard Lyon 1 , 69 008 Lyon , France"},{"name":"Facult\u00e9 de M\u00e9decine Lyon Est, Universit\u00e9 Claude Bernard Lyon 1 , 69 003 Lyon , France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"80","published-online":{"date-parts":[[2022,9,16]]},"reference":[{"issue":"1","key":"2022112511292219400_dgac543-B1","doi-asserted-by":"crossref","first-page":"58","DOI":"10.1186\/s13023-019-1014-8","article-title":"FGF23 and its role in X-linked hypophosphatemia-related morbidity","volume":"14","author":"Beck-Nielsen","year":"2019","journal-title":"Orphanet J Rare Dis"},{"key":"2022112511292219400_dgac543-B2","doi-asserted-by":"crossref","first-page":"153865","DOI":"10.1016\/j.metabol.2019.01.006","article-title":"Physiology of FGF23 and overview of genetic diseases associated with renal phosphate 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