{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,15]],"date-time":"2026-06-15T03:40:15Z","timestamp":1781494815096,"version":"3.54.1"},"reference-count":22,"publisher":"The Endocrine Society","issue":"9","license":[{"start":{"date-parts":[[2025,1,6]],"date-time":"2025-01-06T00:00:00Z","timestamp":1736121600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2025,8,7]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Context<\/jats:title>\n                  <jats:p>Tumor-induced osteomalacia (TIO), a paraneoplastic disorder characterized by renal phosphate wasting, is cured by surgical removal of the culprit tumor. Despite correct localization, some remain refractory to intervention, resulting in substantial long-term medical complications.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Objective<\/jats:title>\n                  <jats:p>We aim to identify risk factors associated with a refractory outcome.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>This is a retrospective cohort of 44 patients with TIO diagnosed from 1998 to 2023 who underwent targeted intervention following successfully localization. Cure was defined as maintenance of normophosphatemia without supplementation for \u22651 month, maintained at last follow-up.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>Twenty-nine patients achieved cure and 15 had a refractory outcome. On univariate Cox regression, the HR for predicting cure was 3.43 (95% CI 1.45-8.11, P = .005) for patients diagnosed after 2013 (compared to before), and that for a negative surgical tumor margin was 2.56 (95% CI 1.20-5.45, P = .015) compared to positive\/unspecified margins. After adjustment for year of diagnosis, tumors originating from soft tissue (HR 2.72 vs bone, 95% CI 1.22-6.09, P = .015) or located outside the spine (HR 0.22 for spine vs nonspine, 95% CI 0.05-0.96, P = .043) had higher chances of cure. Size of tumor, age, gender, or baseline biochemistry including levels of fibroblast growth factor (FGF)23, phosphorus, 1,25-dihydroxyvitamin D, or alkaline phosphatase were not predictive of cure. Postprocedural FGF23 was the best biochemical marker of cure (area under curve 0.899, 95% CI 0.764-1.00, P &amp;lt; .001).<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusion<\/jats:title>\n                  <jats:p>Tumors diagnosed within the past decade with clear resection margins had more favorable prognoses. With regards to tumoral factors, baseline biochemistry was uninformative in predicting cure, while bone and\/or spine localizations were associated with a refractory outcome.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1210\/clinem\/dgae911","type":"journal-article","created":{"date-parts":[[2025,1,6]],"date-time":"2025-01-06T10:31:33Z","timestamp":1736159493000},"page":"2573-2583","source":"Crossref","is-referenced-by-count":2,"title":["Prognostic Factors for Refractory Outcome in Localizing TIO: Experience in a Tertiary Center"],"prefix":"10.1210","volume":"110","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5206-5477","authenticated-orcid":false,"given":"Caroline Wei Shan","family":"Hoong","sequence":"first","affiliation":[{"name":"Division of Endocrinology and Metabolism, Department of Medicine, Mayo Clinic Rochester , Rochester, MN 55905 ,","place":["USA"]},{"name":"Division of Endocrinology, Woodlands Health, National Healthcare Group , Singapore 737628,","place":["Singapore"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1576-1534","authenticated-orcid":false,"given":"Jad G","family":"Sfeir","sequence":"additional","affiliation":[{"name":"Division of Endocrinology and Metabolism, Department of Medicine, Mayo Clinic Rochester , Rochester, MN 55905 ,","place":["USA"]},{"name":"Robert and Arlene Kogod Center on Aging, Mayo Clinic Rochester , Rochester, MN 55905 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2147-0891","authenticated-orcid":false,"given":"Peter","family":"Tebben","sequence":"additional","affiliation":[{"name":"Departments of Internal Medicine and Pediatrics, Section of Endocrinology, Yale School of Medicine , New Haven, CT 06520 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3801-9546","authenticated-orcid":false,"given":"Bart Lyman","family":"Clarke","sequence":"additional","affiliation":[{"name":"Division of Endocrinology and Metabolism, Department of Medicine, Mayo Clinic Rochester , Rochester, MN 55905 ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"80","published-online":{"date-parts":[[2025,1,6]]},"reference":[{"issue":"2","key":"2025081215523280400_dgae911-B1","doi-asserted-by":"crossref","first-page":"e10436","DOI":"10.1002\/jbm4.10436","article-title":"Burden of disease in patients with tumor-induced osteomalacia","volume":"5","author":"Jerkovich","year":"2020","journal-title":"JBMR Plus"},{"issue":"3","key":"2025081215523280400_dgae911-B2","doi-asserted-by":"crossref","first-page":"709","DOI":"10.1007\/s12020-022-03039-2","article-title":"Persistence and recurrence in tumor-induced osteomalacia: a systematic review of the literature and results from a national survey\/case series","volume":"76","author":"Cianferotti","year":"2022","journal-title":"Endocrine"},{"issue":"3","key":"2025081215523280400_dgae911-B3","doi-asserted-by":"crossref","first-page":"469","DOI":"10.1002\/jbmr.3903","article-title":"Nonremission and recurrent tumor-induced osteomalacia: a retrospective study","volume":"35","author":"Li","year":"2020","journal-title":"J Bone Miner Res"},{"key":"2025081215523280400_dgae911-B4","doi-asserted-by":"crossref","first-page":"e397","DOI":"10.1210\/clinem\/dgae183","article-title":"A retrospective cohort of tumor-induced osteomalacia and case series of malignant disease","volume":"110","author":"Hoong","year":"2024","journal-title":"J Clin Endocrinol Metab"},{"issue":"1","key":"2025081215523280400_dgae911-B5","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1097\/00000478-200401000-00001","article-title":"Most osteomalacia-associated mesenchymal tumors are a single histopathologic entity: an analysis of 32 cases and a comprehensive review of the literature","volume":"28","author":"Folpe","year":"2004","journal-title":"Am J Surg Pathol"},{"key":"2025081215523280400_dgae911-B6","doi-asserted-by":"crossref","first-page":"101834","DOI":"10.1016\/j.beem.2023.101834","article-title":"Tumor-induced osteomalacia: an overview","volume":"38","author":"Jadhav","year":"2023","journal-title":"Best Pract Res Clin Endocrinol Metab"},{"issue":"5","key":"2025081215523280400_dgae911-B7","doi-asserted-by":"crossref","first-page":"870","DOI":"10.1016\/j.jos.2020.07.006","article-title":"Surgical margin for phosphaturic mesenchymal tumors in soft tissues: an analysis of the radiological histopathological correlation","volume":"26","author":"Kobayashi","year":"2021","journal-title":"J Orthop Sci"},{"issue":"1","key":"2025081215523280400_dgae911-B8","doi-asserted-by":"crossref","first-page":"43","DOI":"10.1186\/s12891-015-0496-3","article-title":"Surgical treatment of tumor-induced osteomalacia: a retrospective review of 40 cases with extremity tumors","volume":"16","author":"Sun","year":"2015","journal-title":"BMC Musculoskelet Disord"},{"issue":"5","key":"2025081215523280400_dgae911-B9","doi-asserted-by":"crossref","first-page":"900","DOI":"10.1016\/j.jos.2019.02.005","article-title":"Infiltrative nature of tumor-induced osteomalacia lesions in bone: correlation between radiological and histopathological features","volume":"24","author":"Kobayashi","year":"2019","journal-title":"J Orthop Sci"},{"key":"2025081215523280400_dgae911-B10","doi-asserted-by":"crossref","first-page":"902104","DOI":"10.1155\/2014\/902104","article-title":"Role of intraoperative pathology consultation in skeletal tumors and tumor-like lesions","volume":"2014","author":"Bhaker","year":"2014","journal-title":"Sarcoma"},{"issue":"10","key":"2025081215523280400_dgae911-B11","doi-asserted-by":"crossref","first-page":"1044","DOI":"10.1016\/j.jacr.2023.07.017","article-title":"Bone tumor risk stratification and management system: a consensus guideline from the ACR bone reporting and data system committee","volume":"20","author":"Caracciolo","year":"2023","journal-title":"J Am Coll Radiol"},{"key":"2025081215523280400_dgae911-B12","doi-asserted-by":"crossref","first-page":"103108","DOI":"10.1016\/j.amsu.2021.103108","article-title":"Limited utility of intraoperative frozen sections in primary malignant tumours involving long bones\u2014a multicenter analysis of 475 cases","volume":"72","author":"Radhamony","year":"2021","journal-title":"Ann Med Surg (Lond)"},{"issue":"3","key":"2025081215523280400_dgae911-B13","doi-asserted-by":"crossref","first-page":"1024","DOI":"10.1016\/j.athoracsur.2011.04.100","article-title":"Long-term outcomes of en bloc resection of non-small cell lung cancer invading the thoracic inlet and spine","volume":"92","author":"Fadel","year":"2011","journal-title":"Ann Thorac Surg"},{"issue":"7","key":"2025081215523280400_dgae911-B14","doi-asserted-by":"crossref","first-page":"e10178","DOI":"10.1002\/jbm4.10178","article-title":"Successful management of tumor-induced osteomalacia with radiofrequency ablation: a case series","volume":"3","author":"Mishra","year":"2019","journal-title":"JBMR Plus"},{"issue":"4","key":"2025081215523280400_dgae911-B15","doi-asserted-by":"crossref","first-page":"luad086","DOI":"10.1210\/jcemcr\/luad086","article-title":"Thermal ablation for treating tumor-induced osteomalacia in a patient with IV phosphate dependency","volume":"1","author":"van Velsen","year":"2023","journal-title":"JCEM Case Rep"},{"issue":"11","key":"2025081215523280400_dgae911-B16","doi-asserted-by":"crossref","first-page":"1469","DOI":"10.1007\/s00270-022-03356-z","article-title":"Cryoablation for the palliation of painful bone metastasis: a systematic review","volume":"46","author":"Khanmohammadi","year":"2023","journal-title":"Cardiovasc Intervent Radiol"},{"issue":"2","key":"2025081215523280400_dgae911-B17","doi-asserted-by":"crossref","first-page":"278","DOI":"10.4055\/cios19179","article-title":"The efficacy of radiofrequency ablation for bone tumors unsuitable for radical excision","volume":"13","author":"Koo","year":"2021","journal-title":"Clin Orthop Surg"},{"issue":"4","key":"2025081215523280400_dgae911-B18","doi-asserted-by":"crossref","first-page":"627","DOI":"10.1002\/jbmr.4233","article-title":"Burosumab for the treatment of tumor-induced osteomalacia","volume":"36","author":"Jan de Beur","year":"2021","journal-title":"J Bone Miner Res"},{"issue":"8","key":"2025081215523280400_dgae911-B19","doi-asserted-by":"crossref","first-page":"1187","DOI":"10.1359\/jbmr.060507","article-title":"Regulation of C-terminal and intact FGF-23 by dietary phosphate in men and women","volume":"21","author":"Burnett","year":"2006","journal-title":"J Bone Miner Res"},{"issue":"6","key":"2025081215523280400_dgae911-B20","doi-asserted-by":"crossref","first-page":"2374","DOI":"10.1210\/jc.2006-2865","article-title":"Determination of the elimination half-life of fibroblast growth factor-23","volume":"92","author":"Khosravi","year":"2007","journal-title":"J Clin Endocrinol Metab"},{"issue":"1","key":"2025081215523280400_dgae911-B21","doi-asserted-by":"crossref","first-page":"17044","DOI":"10.1038\/nrdp.2017.44","article-title":"Tumour-induced osteomalacia","volume":"3","author":"Minisola","year":"2017","journal-title":"Nat Rev Dis Primers"},{"issue":"6","key":"2025081215523280400_dgae911-B22","doi-asserted-by":"crossref","first-page":"1386","DOI":"10.1002\/jbmr.1881","article-title":"Tumor localization and biochemical response to cure in tumor-induced osteomalacia","volume":"28","author":"Chong","year":"2013","journal-title":"J Bone Miner Res"}],"container-title":["The Journal of Clinical Endocrinology &amp; Metabolism"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/academic.oup.com\/jcem\/advance-article-pdf\/doi\/10.1210\/clinem\/dgae911\/61354116\/dgae911.pdf","content-type":"application\/pdf","content-version":"am","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/jcem\/article-pdf\/110\/9\/2573\/61354116\/dgae911.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"https:\/\/academic.oup.com\/jcem\/article-pdf\/110\/9\/2573\/61354116\/dgae911.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,8,12]],"date-time":"2025-08-12T19:52:42Z","timestamp":1755028362000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/jcem\/article\/110\/9\/2573\/7943416"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,1,6]]},"references-count":22,"journal-issue":{"issue":"9","published-online":{"date-parts":[[2025,1,6]]},"published-print":{"date-parts":[[2025,8,7]]}},"URL":"https:\/\/doi.org\/10.1210\/clinem\/dgae911","relation":{},"ISSN":["0021-972X","1945-7197"],"issn-type":[{"value":"0021-972X","type":"print"},{"value":"1945-7197","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2025,9]]},"published":{"date-parts":[[2025,1,6]]}}}