Tumor-Induced Osteomalacia: Increased Level of FGF-23 in a Patient with a Phosphaturic Mesenchymal Tumor at the Tibia Expressing Periostin
In our case, a 45-year-old male patient had multiple fractures accompanied by hypophosphatemia. FGF-23 levels were significantly increased, and total body magnetic resonance imaging (MRI) revealed a tumor mass located at the distal tibia leading to the diagnosis of tumor-induced osteomalacia (TIO)....
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doaj-6feb6c011440432a8626267a79b86b1b2020-11-24T23:04:53ZengHindawi LimitedCase Reports in Endocrinology2090-65012090-651X2014-01-01201410.1155/2014/729387729387Tumor-Induced Osteomalacia: Increased Level of FGF-23 in a Patient with a Phosphaturic Mesenchymal Tumor at the Tibia Expressing PeriostinAnke H. Hautmann0Josef Schroeder1Peter Wild2Matthias G. Hautmann3Elisabeth Huber4Patrick Hoffstetter5Martin Fleck6Christiane Girlich7Department of Internal Medicine III, Hematology/Internal Oncology, University Hospital of Regensburg, Franz-Josef-Strauß-Allee 11, 93053 Regensburg, GermanyInstitute of Pathology, University Hospital of Regensburg, 93053 Regensburg, GermanyInstitute of Surgical Pathology, University Hospital of Zurich, 8091 Zurich, SwitzerlandDepartment of Radiotherapy, University Hospital of Regensburg, 93053 Regensburg, GermanyInstitute of Pathology, University Hospital of Regensburg, 93053 Regensburg, GermanyInstitute of Radiology, Asklepios Clinic, 93077 Bad Abbach, GermanyDepartment of Internal Medicine I, University Hospital of Regensburg, 93053 Regensburg, GermanyDepartment of General and Geriatric Medicine, Hospital of Barmherzige Brüder, 93049 Regensburg, GermanyIn our case, a 45-year-old male patient had multiple fractures accompanied by hypophosphatemia. FGF-23 levels were significantly increased, and total body magnetic resonance imaging (MRI) revealed a tumor mass located at the distal tibia leading to the diagnosis of tumor-induced osteomalacia (TIO). After resection of the tumor, hypophosphatemia and the increased levels of FGF-23 normalized within a few days. Subsequent microscopic examination and immunohistochemical analysis revealed a phosphaturic mesenchymal tumor mixed connective tissue variant (PMTMCT) showing a positive expression of somatostatin receptor 2A (SSTR2A), CD68, and Periostin. Electron microscopy demonstrated a poorly differentiated mesenchymal tumor with a multifocal giant cell component and evidence of neurosecretory-granules. However, the resected margins showed no tumor-free tissue, and therefore a subsequent postoperative radiotherapy was performed. The patient is still in complete remission after 34 months. Tumor resection of PMTMCTs is the therapy of choice. Subsequent radiotherapy in case of incompletely resected tumors can be an important option to avoid recurrence or metastasis even though this occurs rarely. The prognostic value of expression of Periostin has to be evaluated more precisely in a larger series of patients with TIO.http://dx.doi.org/10.1155/2014/729387 |
collection |
DOAJ |
language |
English |
format |
Article |
sources |
DOAJ |
author |
Anke H. Hautmann Josef Schroeder Peter Wild Matthias G. Hautmann Elisabeth Huber Patrick Hoffstetter Martin Fleck Christiane Girlich |
spellingShingle |
Anke H. Hautmann Josef Schroeder Peter Wild Matthias G. Hautmann Elisabeth Huber Patrick Hoffstetter Martin Fleck Christiane Girlich Tumor-Induced Osteomalacia: Increased Level of FGF-23 in a Patient with a Phosphaturic Mesenchymal Tumor at the Tibia Expressing Periostin Case Reports in Endocrinology |
author_facet |
Anke H. Hautmann Josef Schroeder Peter Wild Matthias G. Hautmann Elisabeth Huber Patrick Hoffstetter Martin Fleck Christiane Girlich |
author_sort |
Anke H. Hautmann |
title |
Tumor-Induced Osteomalacia: Increased Level of FGF-23 in a Patient with a Phosphaturic Mesenchymal Tumor at the Tibia Expressing Periostin |
title_short |
Tumor-Induced Osteomalacia: Increased Level of FGF-23 in a Patient with a Phosphaturic Mesenchymal Tumor at the Tibia Expressing Periostin |
title_full |
Tumor-Induced Osteomalacia: Increased Level of FGF-23 in a Patient with a Phosphaturic Mesenchymal Tumor at the Tibia Expressing Periostin |
title_fullStr |
Tumor-Induced Osteomalacia: Increased Level of FGF-23 in a Patient with a Phosphaturic Mesenchymal Tumor at the Tibia Expressing Periostin |
title_full_unstemmed |
Tumor-Induced Osteomalacia: Increased Level of FGF-23 in a Patient with a Phosphaturic Mesenchymal Tumor at the Tibia Expressing Periostin |
title_sort |
tumor-induced osteomalacia: increased level of fgf-23 in a patient with a phosphaturic mesenchymal tumor at the tibia expressing periostin |
publisher |
Hindawi Limited |
series |
Case Reports in Endocrinology |
issn |
2090-6501 2090-651X |
publishDate |
2014-01-01 |
description |
In our case, a 45-year-old male patient had multiple fractures accompanied by hypophosphatemia. FGF-23 levels were significantly increased, and total body magnetic resonance imaging (MRI) revealed a tumor mass located at the distal tibia leading to the diagnosis of tumor-induced osteomalacia (TIO). After resection of the tumor, hypophosphatemia and the increased levels of FGF-23 normalized within a few days. Subsequent microscopic examination and immunohistochemical analysis revealed a phosphaturic mesenchymal tumor mixed connective tissue variant (PMTMCT) showing a positive expression of somatostatin receptor 2A (SSTR2A), CD68, and Periostin. Electron microscopy demonstrated a poorly differentiated mesenchymal tumor with a multifocal giant cell component and evidence of neurosecretory-granules. However, the resected margins showed no tumor-free tissue, and therefore a subsequent postoperative radiotherapy was performed. The patient is still in complete remission after 34 months. Tumor resection of PMTMCTs is the therapy of choice. Subsequent radiotherapy in case of incompletely resected tumors can be an important option to avoid recurrence or metastasis even though this occurs rarely. The prognostic value of expression of Periostin has to be evaluated more precisely in a larger series of patients with TIO. |
url |
http://dx.doi.org/10.1155/2014/729387 |
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