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Tumour-induced osteomalacia due to an intra-abdominal mesenchymal tumour
A 50-year-man presented with debilitating lower-limb proximal muscle weakness and hip pain since 3 years. Investigations (serum calcium (8.9 mg/dL), serum phosphorus (1.5 mg/dL), serum albumin (40 g/L), parathyroid hormone (116 pg/mL (12.30 pmol/L)), 25(OH)D3 (25.2 ng/mL (63 nmol/L)) 1,25(OH)2 D3 (1...
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Published in: | BMJ case reports 2019-12, Vol.12 (12), p.e231728 |
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description | A 50-year-man presented with debilitating lower-limb proximal muscle weakness and hip pain since 3 years. Investigations (serum calcium (8.9 mg/dL), serum phosphorus (1.5 mg/dL), serum albumin (40 g/L), parathyroid hormone (116 pg/mL (12.30 pmol/L)), 25(OH)D3 (25.2 ng/mL (63 nmol/L)) 1,25(OH)2 D3 (19 pg/mL (45.60 pmol/L)), tubular reabsorption of phosphate of 0.22 and elevated serum fibroblast growth factor 23 (FGF23) (387.7 RU/mL)) were consistent with tumour-induced osteomalacia (TIO). Localisation studies (68Ga DOTATATE positron emission tomography (PET)/CT and 18FDG-PET/CT) did not reveal any lesion. Re-evaluation after 2 and 5 years with 68Ga-DOTANOC PET/CT showed 2×1.4 cm progressively increasing rounded soft tissue enhancing mass close to splenic hilum (SUV max: 26.4). Tumour was resected by laparotomy. Both FGF23 (120 RU/mL on day 3) and serum phosphorus (2.5 mg/dL on day 10) normalised with significant clinical improvement after surgery. Histopathology revealed phosphaturic mesenchymal tumour. Here, we report the first case of intra-abdominal mesenchymal tumour causing TIO diagnosed by serial functional imaging. |
doi_str_mv | 10.1136/bcr-2019-231728 |
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Investigations (serum calcium (8.9 mg/dL), serum phosphorus (1.5 mg/dL), serum albumin (40 g/L), parathyroid hormone (116 pg/mL (12.30 pmol/L)), 25(OH)D3 (25.2 ng/mL (63 nmol/L)) 1,25(OH)2 D3 (19 pg/mL (45.60 pmol/L)), tubular reabsorption of phosphate of 0.22 and elevated serum fibroblast growth factor 23 (FGF23) (387.7 RU/mL)) were consistent with tumour-induced osteomalacia (TIO). Localisation studies (68Ga DOTATATE positron emission tomography (PET)/CT and 18FDG-PET/CT) did not reveal any lesion. Re-evaluation after 2 and 5 years with 68Ga-DOTANOC PET/CT showed 2×1.4 cm progressively increasing rounded soft tissue enhancing mass close to splenic hilum (SUV max: 26.4). Tumour was resected by laparotomy. Both FGF23 (120 RU/mL on day 3) and serum phosphorus (2.5 mg/dL on day 10) normalised with significant clinical improvement after surgery. Histopathology revealed phosphaturic mesenchymal tumour. Here, we report the first case of intra-abdominal mesenchymal tumour causing TIO diagnosed by serial functional imaging.</description><identifier>ISSN: 1757-790X</identifier><identifier>EISSN: 1757-790X</identifier><identifier>DOI: 10.1136/bcr-2019-231728</identifier><identifier>PMID: 31806630</identifier><language>eng</language><publisher>England: BMJ Publishing Group Ltd</publisher><subject>Abdomen ; Abdominal Neoplasms - complications ; Abdominal Neoplasms - diagnosis ; Abdominal Neoplasms - diagnostic imaging ; Abdominal Neoplasms - pathology ; Bones ; calcium and bone ; Case reports ; endocrine cancer ; Fibroblasts ; Growth factors ; Humans ; Localization ; Male ; Middle Aged ; Neuroendocrine tumors ; Osteomalacia - diagnostic imaging ; Osteomalacia - etiology ; Osteomalacia - pathology ; Patients ; Positron Emission Tomography Computed Tomography ; Positron-Emission Tomography ; radiology ; Rare Disease ; Soft Tissue Neoplasms - complications ; Soft Tissue Neoplasms - diagnosis ; Soft Tissue Neoplasms - diagnostic imaging ; Soft Tissue Neoplasms - pathology ; Surgery ; Tomography ; Tumors</subject><ispartof>BMJ case reports, 2019-12, Vol.12 (12), p.e231728</ispartof><rights>BMJ Publishing Group Limited 2019. No commercial re-use. See rights and permissions. Published by BMJ.</rights><rights>2019 BMJ Publishing Group Limited 2019. No commercial re-use. See rights and permissions. Published by BMJ.</rights><rights>BMJ Publishing Group Limited 2019. No commercial re-use. See rights and permissions. Published by BMJ. 2019</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-b492t-572bbf85e56085b39a20edaef6dbc7ac370ed3543f02965085f1ee736a1263b53</citedby><cites>FETCH-LOGICAL-b492t-572bbf85e56085b39a20edaef6dbc7ac370ed3543f02965085f1ee736a1263b53</cites><orcidid>0000-0002-8927-9217</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC6904179/pdf/$$EPDF$$P50$$Gpubmedcentral$$H</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC6904179/$$EHTML$$P50$$Gpubmedcentral$$H</linktohtml><link.rule.ids>230,314,727,780,784,885,27924,27925,53791,53793</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/31806630$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Krishnappa, Brijesh</creatorcontrib><creatorcontrib>Jadhav, Swati Ramteke</creatorcontrib><creatorcontrib>Lila, Anurag R</creatorcontrib><creatorcontrib>Bandgar, Tushar R</creatorcontrib><title>Tumour-induced osteomalacia due to an intra-abdominal mesenchymal tumour</title><title>BMJ case reports</title><addtitle>BMJ Case Rep</addtitle><addtitle>BMJ Case Rep</addtitle><description>A 50-year-man presented with debilitating lower-limb proximal muscle weakness and hip pain since 3 years. Investigations (serum calcium (8.9 mg/dL), serum phosphorus (1.5 mg/dL), serum albumin (40 g/L), parathyroid hormone (116 pg/mL (12.30 pmol/L)), 25(OH)D3 (25.2 ng/mL (63 nmol/L)) 1,25(OH)2 D3 (19 pg/mL (45.60 pmol/L)), tubular reabsorption of phosphate of 0.22 and elevated serum fibroblast growth factor 23 (FGF23) (387.7 RU/mL)) were consistent with tumour-induced osteomalacia (TIO). Localisation studies (68Ga DOTATATE positron emission tomography (PET)/CT and 18FDG-PET/CT) did not reveal any lesion. Re-evaluation after 2 and 5 years with 68Ga-DOTANOC PET/CT showed 2×1.4 cm progressively increasing rounded soft tissue enhancing mass close to splenic hilum (SUV max: 26.4). Tumour was resected by laparotomy. Both FGF23 (120 RU/mL on day 3) and serum phosphorus (2.5 mg/dL on day 10) normalised with significant clinical improvement after surgery. Histopathology revealed phosphaturic mesenchymal tumour. Here, we report the first case of intra-abdominal mesenchymal tumour causing TIO diagnosed by serial functional imaging.</description><subject>Abdomen</subject><subject>Abdominal Neoplasms - complications</subject><subject>Abdominal Neoplasms - diagnosis</subject><subject>Abdominal Neoplasms - diagnostic imaging</subject><subject>Abdominal Neoplasms - pathology</subject><subject>Bones</subject><subject>calcium and bone</subject><subject>Case reports</subject><subject>endocrine cancer</subject><subject>Fibroblasts</subject><subject>Growth factors</subject><subject>Humans</subject><subject>Localization</subject><subject>Male</subject><subject>Middle Aged</subject><subject>Neuroendocrine tumors</subject><subject>Osteomalacia - diagnostic imaging</subject><subject>Osteomalacia - etiology</subject><subject>Osteomalacia - pathology</subject><subject>Patients</subject><subject>Positron Emission Tomography Computed Tomography</subject><subject>Positron-Emission Tomography</subject><subject>radiology</subject><subject>Rare Disease</subject><subject>Soft Tissue Neoplasms - complications</subject><subject>Soft Tissue Neoplasms - diagnosis</subject><subject>Soft Tissue Neoplasms - diagnostic imaging</subject><subject>Soft Tissue Neoplasms - pathology</subject><subject>Surgery</subject><subject>Tomography</subject><subject>Tumors</subject><issn>1757-790X</issn><issn>1757-790X</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2019</creationdate><recordtype>article</recordtype><recordid>eNqNkUtLxDAUhYMoKuOs3UnBjSh18miSdiOI-IIBNyO4C0l6qx3aRptW8N-bWh1HQTCbJOTLuedwENon-JQQJmbGtjHFJIspI5KmG2iXSC5jmeGHzbXzDpp6v8RhMZKkCdtGO4ykWAiGd9HNoq9d38Zlk_cW8sj5DlytK21LHeU9RJ2LdBOVTdfqWJvc1WWjq6gGD419egtk1H0o7KGtQlcepp_7BN1fXS4ubuL53fXtxfk8NklGu5hLakyRcuACp9ywTFMMuYZC5MZKbZkMV8YTVmCaCR6YggBIJjShghnOJuhs1H3uTQ25hcFZpZ7bstbtm3K6VD9fmvJJPbpXJTKcEJkFgaNPgda99OA7VZfeQlXpBlzvFWWUSk4yOcw6_IUuQ9KQf6CSAEkiZKBmI2Vb530LxcoMwWooSoWi1FCUGosKPw7WM6z4r1oCcDwCpl7-Q-3kG14Z_It-B8F6qZM</recordid><startdate>20191204</startdate><enddate>20191204</enddate><creator>Krishnappa, Brijesh</creator><creator>Jadhav, Swati Ramteke</creator><creator>Lila, Anurag R</creator><creator>Bandgar, Tushar R</creator><general>BMJ Publishing Group Ltd</general><general>BMJ Publishing Group LTD</general><general>BMJ Publishing Group</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7RV</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>BENPR</scope><scope>BTHHO</scope><scope>CCPQU</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>K9.</scope><scope>KB0</scope><scope>M0S</scope><scope>M1P</scope><scope>NAPCQ</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>7X8</scope><scope>5PM</scope><orcidid>https://orcid.org/0000-0002-8927-9217</orcidid></search><sort><creationdate>20191204</creationdate><title>Tumour-induced osteomalacia due to an intra-abdominal mesenchymal tumour</title><author>Krishnappa, Brijesh ; Jadhav, Swati Ramteke ; Lila, Anurag R ; Bandgar, Tushar R</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-b492t-572bbf85e56085b39a20edaef6dbc7ac370ed3543f02965085f1ee736a1263b53</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2019</creationdate><topic>Abdomen</topic><topic>Abdominal Neoplasms - complications</topic><topic>Abdominal Neoplasms - diagnosis</topic><topic>Abdominal Neoplasms - diagnostic imaging</topic><topic>Abdominal Neoplasms - pathology</topic><topic>Bones</topic><topic>calcium and bone</topic><topic>Case reports</topic><topic>endocrine cancer</topic><topic>Fibroblasts</topic><topic>Growth factors</topic><topic>Humans</topic><topic>Localization</topic><topic>Male</topic><topic>Middle Aged</topic><topic>Neuroendocrine tumors</topic><topic>Osteomalacia - diagnostic imaging</topic><topic>Osteomalacia - etiology</topic><topic>Osteomalacia - pathology</topic><topic>Patients</topic><topic>Positron Emission Tomography Computed Tomography</topic><topic>Positron-Emission Tomography</topic><topic>radiology</topic><topic>Rare Disease</topic><topic>Soft Tissue Neoplasms - complications</topic><topic>Soft Tissue Neoplasms - diagnosis</topic><topic>Soft Tissue Neoplasms - diagnostic imaging</topic><topic>Soft Tissue Neoplasms - pathology</topic><topic>Surgery</topic><topic>Tomography</topic><topic>Tumors</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Krishnappa, Brijesh</creatorcontrib><creatorcontrib>Jadhav, Swati Ramteke</creatorcontrib><creatorcontrib>Lila, Anurag R</creatorcontrib><creatorcontrib>Bandgar, Tushar R</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Nursing & Allied Health Database</collection><collection>ProQuest - Health & Medical Complete保健、医学与药学数据库</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>ProQuest Central (Alumni)</collection><collection>ProQuest Central</collection><collection>ProQuest Central</collection><collection>BMJ Journals</collection><collection>ProQuest One Community College</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Nursing & Allied Health Database (Alumni Edition)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>Nursing & Allied Health Premium</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>BMJ case reports</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Krishnappa, Brijesh</au><au>Jadhav, Swati Ramteke</au><au>Lila, Anurag R</au><au>Bandgar, Tushar R</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Tumour-induced osteomalacia due to an intra-abdominal mesenchymal tumour</atitle><jtitle>BMJ case reports</jtitle><stitle>BMJ Case Rep</stitle><addtitle>BMJ Case Rep</addtitle><date>2019-12-04</date><risdate>2019</risdate><volume>12</volume><issue>12</issue><spage>e231728</spage><pages>e231728-</pages><issn>1757-790X</issn><eissn>1757-790X</eissn><abstract>A 50-year-man presented with debilitating lower-limb proximal muscle weakness and hip pain since 3 years. Investigations (serum calcium (8.9 mg/dL), serum phosphorus (1.5 mg/dL), serum albumin (40 g/L), parathyroid hormone (116 pg/mL (12.30 pmol/L)), 25(OH)D3 (25.2 ng/mL (63 nmol/L)) 1,25(OH)2 D3 (19 pg/mL (45.60 pmol/L)), tubular reabsorption of phosphate of 0.22 and elevated serum fibroblast growth factor 23 (FGF23) (387.7 RU/mL)) were consistent with tumour-induced osteomalacia (TIO). Localisation studies (68Ga DOTATATE positron emission tomography (PET)/CT and 18FDG-PET/CT) did not reveal any lesion. Re-evaluation after 2 and 5 years with 68Ga-DOTANOC PET/CT showed 2×1.4 cm progressively increasing rounded soft tissue enhancing mass close to splenic hilum (SUV max: 26.4). Tumour was resected by laparotomy. Both FGF23 (120 RU/mL on day 3) and serum phosphorus (2.5 mg/dL on day 10) normalised with significant clinical improvement after surgery. Histopathology revealed phosphaturic mesenchymal tumour. Here, we report the first case of intra-abdominal mesenchymal tumour causing TIO diagnosed by serial functional imaging.</abstract><cop>England</cop><pub>BMJ Publishing Group Ltd</pub><pmid>31806630</pmid><doi>10.1136/bcr-2019-231728</doi><orcidid>https://orcid.org/0000-0002-8927-9217</orcidid><oa>free_for_read</oa></addata></record> |
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subjects | Abdomen Abdominal Neoplasms - complications Abdominal Neoplasms - diagnosis Abdominal Neoplasms - diagnostic imaging Abdominal Neoplasms - pathology Bones calcium and bone Case reports endocrine cancer Fibroblasts Growth factors Humans Localization Male Middle Aged Neuroendocrine tumors Osteomalacia - diagnostic imaging Osteomalacia - etiology Osteomalacia - pathology Patients Positron Emission Tomography Computed Tomography Positron-Emission Tomography radiology Rare Disease Soft Tissue Neoplasms - complications Soft Tissue Neoplasms - diagnosis Soft Tissue Neoplasms - diagnostic imaging Soft Tissue Neoplasms - pathology Surgery Tomography Tumors |
title | Tumour-induced osteomalacia due to an intra-abdominal mesenchymal tumour |
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