Caroline Wei Shan Hoong, Jad G Sfeir, Peter Tebben, Bart Lyman Clarke
{"title":"Prognostic factors for refractory outcome in localizing TIO: experience in a tertiary center.","authors":"Caroline Wei Shan Hoong, Jad G Sfeir, Peter Tebben, Bart Lyman Clarke","doi":"10.1210/clinem/dgae911","DOIUrl":null,"url":null,"abstract":"<p><strong>Context: </strong>TIO, a paraneoplastic disorder characterised by renal phosphate wasting, is cured by surgical removal of the culprit tumour. Despite correct localization, some remain refractory to intervention, resulting in substantial long-term medical complications.</p><p><strong>Aim: </strong>We aim to identify risk factors associated with a refractory outcome.</p><p><strong>Methods: </strong>This is a retrospective cohort of 44 TIO patients diagnosed from 1998-2023 who underwent targeted intervention following successfully localization. Cure was defined as maintenance of normophosphatemia without supplementation for >1 month, maintained at last follow-up.</p><p><strong>Results: </strong>29 patients achieved cure and 15 had a refractory outcome. On univariate cox regression, HR for predicting cure was 3.43 (95%CI 1.45-8.11, p=0.005) 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=0.015) compared to positive/unspecified margins. After adjustment for year of diagnosis, tumors originating from soft tissue (HR 2.72 versus bone, 95%CI 1.22-6.09, p=0.015) or located outside the spine (HR 0.22 for spine versus non-spine, 95%CI 0.05-0.96, p=0.043) had higher chances of cure. Size of tumor, age, gender, or baseline biochemistry including levels of FGF23, phosphorus, 1,25(OH)2D or ALP were not predictive of cure. Post-procedural FGF23 was the best biochemical marker of cure (AUC 0.899, 95%CI 0.764-1.00, p < 0.001).</p><p><strong>Conclusion: </strong>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.</p>","PeriodicalId":50238,"journal":{"name":"Journal of Clinical Endocrinology & Metabolism","volume":" ","pages":""},"PeriodicalIF":5.0000,"publicationDate":"2025-01-06","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal of Clinical Endocrinology & Metabolism","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1210/clinem/dgae911","RegionNum":2,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q1","JCRName":"ENDOCRINOLOGY & METABOLISM","Score":null,"Total":0}
引用次数: 0
Abstract
Context: TIO, a paraneoplastic disorder characterised by renal phosphate wasting, is cured by surgical removal of the culprit tumour. Despite correct localization, some remain refractory to intervention, resulting in substantial long-term medical complications.
Aim: We aim to identify risk factors associated with a refractory outcome.
Methods: This is a retrospective cohort of 44 TIO patients diagnosed from 1998-2023 who underwent targeted intervention following successfully localization. Cure was defined as maintenance of normophosphatemia without supplementation for >1 month, maintained at last follow-up.
Results: 29 patients achieved cure and 15 had a refractory outcome. On univariate cox regression, HR for predicting cure was 3.43 (95%CI 1.45-8.11, p=0.005) 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=0.015) compared to positive/unspecified margins. After adjustment for year of diagnosis, tumors originating from soft tissue (HR 2.72 versus bone, 95%CI 1.22-6.09, p=0.015) or located outside the spine (HR 0.22 for spine versus non-spine, 95%CI 0.05-0.96, p=0.043) had higher chances of cure. Size of tumor, age, gender, or baseline biochemistry including levels of FGF23, phosphorus, 1,25(OH)2D or ALP were not predictive of cure. Post-procedural FGF23 was the best biochemical marker of cure (AUC 0.899, 95%CI 0.764-1.00, p < 0.001).
Conclusion: 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.
期刊介绍:
The Journal of Clinical Endocrinology & Metabolism is the world"s leading peer-reviewed journal for endocrine clinical research and cutting edge clinical practice reviews. Each issue provides the latest in-depth coverage of new developments enhancing our understanding, diagnosis and treatment of endocrine and metabolic disorders. Regular features of special interest to endocrine consultants include clinical trials, clinical reviews, clinical practice guidelines, case seminars, and controversies in clinical endocrinology, as well as original reports of the most important advances in patient-oriented endocrine and metabolic research. According to the latest Thomson Reuters Journal Citation Report, JCE&M articles were cited 64,185 times in 2008.