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Oncogenic Osteomalacia Caused by a Phosphaturic Mesenchymal Tumor of the Oral Cavity: A Case Report

We report a case of oncogenic osteomalacia associated with a phosphaturic mesenchymal tumor in a 31-year-old woman. She was presented with severe generalized bone and muscle pain and was restricted to bed. She lost 20cm in height over the 8 years since she had first noticed a pain in her thigh. A wa...

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Autores principales: Yang, In Myung, Park, Yong Koo, Hyun, Yong Jun, Kim, Deog Yoon, Woo, Jeong Taek, Kim, Sung Woon, Kim, Jin Woo, Kim, Young Seol, Choi, Young Kil
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Korean Association of Internal Medicine 1997
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4531958/
https://www.ncbi.nlm.nih.gov/pubmed/9159046
http://dx.doi.org/10.3904/kjim.1997.12.1.89
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author Yang, In Myung
Park, Yong Koo
Hyun, Yong Jun
Kim, Deog Yoon
Woo, Jeong Taek
Kim, Sung Woon
Kim, Jin Woo
Kim, Young Seol
Choi, Young Kil
author_facet Yang, In Myung
Park, Yong Koo
Hyun, Yong Jun
Kim, Deog Yoon
Woo, Jeong Taek
Kim, Sung Woon
Kim, Jin Woo
Kim, Young Seol
Choi, Young Kil
author_sort Yang, In Myung
collection PubMed
description We report a case of oncogenic osteomalacia associated with a phosphaturic mesenchymal tumor in a 31-year-old woman. She was presented with severe generalized bone and muscle pain and was restricted to bed. She lost 20cm in height over the 8 years since she had first noticed a pain in her thigh. A walnut-sized, hard, soft tissue tumor was found very easily beside her lower molar teeth. Radiologic examination revealed a remarkable decrease in bone density and multiple pathologic fractures of spine, femur and phalangeal bones. Severe hypophosphatemia, hyperphosphaturia, low plasma 1,25-dihydroxyvitamin D(3) level and high plasma PTH level were disclosed at presentation. Histomorphometric examination revealed an extensive area of unmineralized osteoid and little mineralizing activity. A pharmacologic dose of 1α-hydroxyvitamin D(3) or 1,25-dihydroxyvitamin D(3) slightly increased the serum phosphate level and renal tubular reabsorption of phosphate, and slightly decreased plasma PTH level without any symptomatic improvement. Histologic examination of the tumor revealed a mixed connective tissue tumor that consisted of central woveh bones and surrounding primitive spindle cells with prominent vascularities. After removal of the tumor, all biochemical, hormonal and radiologic abnormalities disappeared with remarkable symptomatic improvement.
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spelling pubmed-45319582015-10-02 Oncogenic Osteomalacia Caused by a Phosphaturic Mesenchymal Tumor of the Oral Cavity: A Case Report Yang, In Myung Park, Yong Koo Hyun, Yong Jun Kim, Deog Yoon Woo, Jeong Taek Kim, Sung Woon Kim, Jin Woo Kim, Young Seol Choi, Young Kil Korean J Intern Med Case Report We report a case of oncogenic osteomalacia associated with a phosphaturic mesenchymal tumor in a 31-year-old woman. She was presented with severe generalized bone and muscle pain and was restricted to bed. She lost 20cm in height over the 8 years since she had first noticed a pain in her thigh. A walnut-sized, hard, soft tissue tumor was found very easily beside her lower molar teeth. Radiologic examination revealed a remarkable decrease in bone density and multiple pathologic fractures of spine, femur and phalangeal bones. Severe hypophosphatemia, hyperphosphaturia, low plasma 1,25-dihydroxyvitamin D(3) level and high plasma PTH level were disclosed at presentation. Histomorphometric examination revealed an extensive area of unmineralized osteoid and little mineralizing activity. A pharmacologic dose of 1α-hydroxyvitamin D(3) or 1,25-dihydroxyvitamin D(3) slightly increased the serum phosphate level and renal tubular reabsorption of phosphate, and slightly decreased plasma PTH level without any symptomatic improvement. Histologic examination of the tumor revealed a mixed connective tissue tumor that consisted of central woveh bones and surrounding primitive spindle cells with prominent vascularities. After removal of the tumor, all biochemical, hormonal and radiologic abnormalities disappeared with remarkable symptomatic improvement. Korean Association of Internal Medicine 1997-01 /pmc/articles/PMC4531958/ /pubmed/9159046 http://dx.doi.org/10.3904/kjim.1997.12.1.89 Text en Copyright © 1997 The Korean Association of Internal Medicine This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0/) which permits unrestricted noncommercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Yang, In Myung
Park, Yong Koo
Hyun, Yong Jun
Kim, Deog Yoon
Woo, Jeong Taek
Kim, Sung Woon
Kim, Jin Woo
Kim, Young Seol
Choi, Young Kil
Oncogenic Osteomalacia Caused by a Phosphaturic Mesenchymal Tumor of the Oral Cavity: A Case Report
title Oncogenic Osteomalacia Caused by a Phosphaturic Mesenchymal Tumor of the Oral Cavity: A Case Report
title_full Oncogenic Osteomalacia Caused by a Phosphaturic Mesenchymal Tumor of the Oral Cavity: A Case Report
title_fullStr Oncogenic Osteomalacia Caused by a Phosphaturic Mesenchymal Tumor of the Oral Cavity: A Case Report
title_full_unstemmed Oncogenic Osteomalacia Caused by a Phosphaturic Mesenchymal Tumor of the Oral Cavity: A Case Report
title_short Oncogenic Osteomalacia Caused by a Phosphaturic Mesenchymal Tumor of the Oral Cavity: A Case Report
title_sort oncogenic osteomalacia caused by a phosphaturic mesenchymal tumor of the oral cavity: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4531958/
https://www.ncbi.nlm.nih.gov/pubmed/9159046
http://dx.doi.org/10.3904/kjim.1997.12.1.89
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