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Phosphaturic mesenchymal tumors among elderly patients: a case report and review of literature

Phosphaturic mesenchymal tumor (PMT) represents a rare cause of osteomalacia. The clinical signs and symptoms are vague and these lead to diagnosis delay. In the presence of hypophosphatemia and relatively high urine phosphate excretion, this entity should be taken into consideration in the deferent...

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Autores principales: Adnan, Zaina, Nikomarov, David, Weiler-Sagie, Michal, Roguin Maor, Noga
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Bioscientifica Ltd 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6499915/
https://www.ncbi.nlm.nih.gov/pubmed/31051470
http://dx.doi.org/10.1530/EDM-18-01396
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author Adnan, Zaina
Nikomarov, David
Weiler-Sagie, Michal
Roguin Maor, Noga
author_facet Adnan, Zaina
Nikomarov, David
Weiler-Sagie, Michal
Roguin Maor, Noga
author_sort Adnan, Zaina
collection PubMed
description Phosphaturic mesenchymal tumor (PMT) represents a rare cause of osteomalacia. The clinical signs and symptoms are vague and these lead to diagnosis delay. In the presence of hypophosphatemia and relatively high urine phosphate excretion, this entity should be taken into consideration in the deferential diagnosis of osteomalacia. In the present article, we report 81-year-old man presented to our clinic for evaluation due to osteopenia. His laboratory results disclosed hypophosphatemia, relatively increased urine phosphate excretion and increased level of intact fibroblast growth factor 23 (FGF23). A (68)Gallium DOTATATE PET/CT revealed pathological uptake in the upper aspect of the left shoulder adjacent to the coracoid process. For suspected PMT a wide resection of the tumor was performed and pathological findings were consistent for PMT. Laboratory tests were normalized postoperatively. Reviewing the literature, we had identified 33 reported cases of PMTs among elderly patients age ≥70 years. Unlike previously reported data, where tumors predominantly localized in the lower extremities and pelvis, our search disclosed a high rate of tumor localization (10 cases – 33.3%) in the head with equal number of tumors (14 cases – 42.4%) localized in the head and upper extremity as well as in pelvis and lower extremity. The present case describes unique tumor localization in an elderly patient and our literature search demonstrated for the first time a high rate of tumor localization in the head among this group of patients. LEARNING POINTS: PMTs represent a rare entity that should be considered in the differential diagnosis of elderly patients presented with persistent hypophosphatemia. Unlike previously reported data, head and neck tumor localization is frequent among elderly patients. (68)Gallium-conjugated somatostatin peptide analogs, such as (68)Ga-DOTATATE PET/CT demonstrated the greatest sensitivity and specificity for tumor localization in patients with phosphaturic mesenchymal tumors (PMTs). Wide tumor resection using intraoperative ultrasound is of major importance in order to ensure long-term cure.
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spelling pubmed-64999152019-05-07 Phosphaturic mesenchymal tumors among elderly patients: a case report and review of literature Adnan, Zaina Nikomarov, David Weiler-Sagie, Michal Roguin Maor, Noga Endocrinol Diabetes Metab Case Rep Unique/Unexpected Symptoms or Presentations of a Disease Phosphaturic mesenchymal tumor (PMT) represents a rare cause of osteomalacia. The clinical signs and symptoms are vague and these lead to diagnosis delay. In the presence of hypophosphatemia and relatively high urine phosphate excretion, this entity should be taken into consideration in the deferential diagnosis of osteomalacia. In the present article, we report 81-year-old man presented to our clinic for evaluation due to osteopenia. His laboratory results disclosed hypophosphatemia, relatively increased urine phosphate excretion and increased level of intact fibroblast growth factor 23 (FGF23). A (68)Gallium DOTATATE PET/CT revealed pathological uptake in the upper aspect of the left shoulder adjacent to the coracoid process. For suspected PMT a wide resection of the tumor was performed and pathological findings were consistent for PMT. Laboratory tests were normalized postoperatively. Reviewing the literature, we had identified 33 reported cases of PMTs among elderly patients age ≥70 years. Unlike previously reported data, where tumors predominantly localized in the lower extremities and pelvis, our search disclosed a high rate of tumor localization (10 cases – 33.3%) in the head with equal number of tumors (14 cases – 42.4%) localized in the head and upper extremity as well as in pelvis and lower extremity. The present case describes unique tumor localization in an elderly patient and our literature search demonstrated for the first time a high rate of tumor localization in the head among this group of patients. LEARNING POINTS: PMTs represent a rare entity that should be considered in the differential diagnosis of elderly patients presented with persistent hypophosphatemia. Unlike previously reported data, head and neck tumor localization is frequent among elderly patients. (68)Gallium-conjugated somatostatin peptide analogs, such as (68)Ga-DOTATATE PET/CT demonstrated the greatest sensitivity and specificity for tumor localization in patients with phosphaturic mesenchymal tumors (PMTs). Wide tumor resection using intraoperative ultrasound is of major importance in order to ensure long-term cure. Bioscientifica Ltd 2019-05-03 /pmc/articles/PMC6499915/ /pubmed/31051470 http://dx.doi.org/10.1530/EDM-18-01396 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 Unique/Unexpected Symptoms or Presentations of a Disease
Adnan, Zaina
Nikomarov, David
Weiler-Sagie, Michal
Roguin Maor, Noga
Phosphaturic mesenchymal tumors among elderly patients: a case report and review of literature
title Phosphaturic mesenchymal tumors among elderly patients: a case report and review of literature
title_full Phosphaturic mesenchymal tumors among elderly patients: a case report and review of literature
title_fullStr Phosphaturic mesenchymal tumors among elderly patients: a case report and review of literature
title_full_unstemmed Phosphaturic mesenchymal tumors among elderly patients: a case report and review of literature
title_short Phosphaturic mesenchymal tumors among elderly patients: a case report and review of literature
title_sort phosphaturic mesenchymal tumors among elderly patients: a case report and review of literature
topic Unique/Unexpected Symptoms or Presentations of a Disease
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6499915/
https://www.ncbi.nlm.nih.gov/pubmed/31051470
http://dx.doi.org/10.1530/EDM-18-01396
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