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Tumor-induced osteomalacia: experience from three tertiary care centers in India

Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome characterized by recalcitrant hypophosphatemia. Reports from the Indian subcontinent are scarce, with most being single center experiences involving few patients. Herein, we conducted a retrospective analysis of 30 patients of TIO di...

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Autores principales: Pal, Rimesh, Bhadada, Sanjay Kumar, Singhare, Awesh, Bhansali, Anil, Kamalanathan, Sadishkumar, Chadha, Manoj, Chauhan, Phulrenu, Sood, Ashwani, Dhiman, Vandana, Sharma, Dinesh Chandra, Saikia, Uma Nahar, Chatterjee, Debajyoti, Agashe, Vikas
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Bioscientifica Ltd 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6410764/
https://www.ncbi.nlm.nih.gov/pubmed/30726771
http://dx.doi.org/10.1530/EC-18-0552
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author Pal, Rimesh
Bhadada, Sanjay Kumar
Singhare, Awesh
Bhansali, Anil
Kamalanathan, Sadishkumar
Chadha, Manoj
Chauhan, Phulrenu
Sood, Ashwani
Dhiman, Vandana
Sharma, Dinesh Chandra
Saikia, Uma Nahar
Chatterjee, Debajyoti
Agashe, Vikas
author_facet Pal, Rimesh
Bhadada, Sanjay Kumar
Singhare, Awesh
Bhansali, Anil
Kamalanathan, Sadishkumar
Chadha, Manoj
Chauhan, Phulrenu
Sood, Ashwani
Dhiman, Vandana
Sharma, Dinesh Chandra
Saikia, Uma Nahar
Chatterjee, Debajyoti
Agashe, Vikas
author_sort Pal, Rimesh
collection PubMed
description Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome characterized by recalcitrant hypophosphatemia. Reports from the Indian subcontinent are scarce, with most being single center experiences involving few patients. Herein, we conducted a retrospective analysis of 30 patients of TIO diagnosed at three tertiary care hospitals in India. Patients with persistent hypophosphatemia (despite correction of hypovitaminosis D), normocalcemia, elevated alkaline phosphatase, low TmP/GFR and elevated or ‘inappropriately normal’ FGF23 levels were labeled as having TIO. They were sequentially subjected to functional followed by anatomical imaging. Patients with a well-localized tumor underwent excision; others were put on phosphorous and calcitriol supplementation. The mean age at presentation was 39.6 years with female:male ratio of 3:2. Bone pain (83.3%) and proximal myopathy (70%) were the chief complaints; 40% of cases had fractures. The mean delay in diagnosis was 3.8 years. Tumors were clinically detectable in four patients (13.3%). The mean serum phosphate was 0.50 mmol/L with a median serum FGF23 level of 518 RU/mL. Somatostatin receptor-based scintigraphy was found to be superior to FDG-PET in tumor localization. Lower extremities were the most common site of the tumor (72%). Tumor size was positively correlated with serum FGF23 levels. Twenty-two patients underwent tumor resection and 16 of them had phosphaturic mesenchymal tumors. Surgical excision led to cure in 72.7% of patients whereas disease persistence and disease recurrence were seen in 18.2% and 9.1% of cases, respectively. At the last follow-up, serum phosphate in the surgically treated group was significantly higher than in the medically managed group.
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spelling pubmed-64107642019-03-14 Tumor-induced osteomalacia: experience from three tertiary care centers in India Pal, Rimesh Bhadada, Sanjay Kumar Singhare, Awesh Bhansali, Anil Kamalanathan, Sadishkumar Chadha, Manoj Chauhan, Phulrenu Sood, Ashwani Dhiman, Vandana Sharma, Dinesh Chandra Saikia, Uma Nahar Chatterjee, Debajyoti Agashe, Vikas Endocr Connect Research Tumor-induced osteomalacia (TIO) is a rare paraneoplastic syndrome characterized by recalcitrant hypophosphatemia. Reports from the Indian subcontinent are scarce, with most being single center experiences involving few patients. Herein, we conducted a retrospective analysis of 30 patients of TIO diagnosed at three tertiary care hospitals in India. Patients with persistent hypophosphatemia (despite correction of hypovitaminosis D), normocalcemia, elevated alkaline phosphatase, low TmP/GFR and elevated or ‘inappropriately normal’ FGF23 levels were labeled as having TIO. They were sequentially subjected to functional followed by anatomical imaging. Patients with a well-localized tumor underwent excision; others were put on phosphorous and calcitriol supplementation. The mean age at presentation was 39.6 years with female:male ratio of 3:2. Bone pain (83.3%) and proximal myopathy (70%) were the chief complaints; 40% of cases had fractures. The mean delay in diagnosis was 3.8 years. Tumors were clinically detectable in four patients (13.3%). The mean serum phosphate was 0.50 mmol/L with a median serum FGF23 level of 518 RU/mL. Somatostatin receptor-based scintigraphy was found to be superior to FDG-PET in tumor localization. Lower extremities were the most common site of the tumor (72%). Tumor size was positively correlated with serum FGF23 levels. Twenty-two patients underwent tumor resection and 16 of them had phosphaturic mesenchymal tumors. Surgical excision led to cure in 72.7% of patients whereas disease persistence and disease recurrence were seen in 18.2% and 9.1% of cases, respectively. At the last follow-up, serum phosphate in the surgically treated group was significantly higher than in the medically managed group. Bioscientifica Ltd 2019-01-28 /pmc/articles/PMC6410764/ /pubmed/30726771 http://dx.doi.org/10.1530/EC-18-0552 Text en © 2019 The authors http://creativecommons.org/licenses/by-nc-nd/4.0/ This work is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License. (http://creativecommons.org/licenses/by-nc-nd/4.0/)
spellingShingle Research
Pal, Rimesh
Bhadada, Sanjay Kumar
Singhare, Awesh
Bhansali, Anil
Kamalanathan, Sadishkumar
Chadha, Manoj
Chauhan, Phulrenu
Sood, Ashwani
Dhiman, Vandana
Sharma, Dinesh Chandra
Saikia, Uma Nahar
Chatterjee, Debajyoti
Agashe, Vikas
Tumor-induced osteomalacia: experience from three tertiary care centers in India
title Tumor-induced osteomalacia: experience from three tertiary care centers in India
title_full Tumor-induced osteomalacia: experience from three tertiary care centers in India
title_fullStr Tumor-induced osteomalacia: experience from three tertiary care centers in India
title_full_unstemmed Tumor-induced osteomalacia: experience from three tertiary care centers in India
title_short Tumor-induced osteomalacia: experience from three tertiary care centers in India
title_sort tumor-induced osteomalacia: experience from three tertiary care centers in india
topic Research
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6410764/
https://www.ncbi.nlm.nih.gov/pubmed/30726771
http://dx.doi.org/10.1530/EC-18-0552
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