Phosphaturic mesenchymal tumor, nonphosphaturic variant, causing fatal pulmonary metastasis
Summary Phosphaturic mesenchymal tumors of the mixed connective tissue type (PMT-MCTs) are rare neoplasms, most of which are benign and cause tumor-induced osteomalacia because of overproduction of a phosphaturic hormone, fibroblast growth factor 23 (FGF23). This entity may have been unrecognized or...
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| Veröffentlicht in: | Human pathology 2013-11, Vol.44 (11), p.2614-2618 |
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| creator | Uchihashi, Kazuyoshi, MD, PhD Nishijima-Matsunobu, Aki, MD, PhD Matsuyama, Atsuji, MD, PhD Yamasaki, Fumio, MD, PhD Tanabe, Tsuyoshi, MD Uemura, Tetsuji, MD, PhD Aragane, Naoko, MD, PhD Yakushiji, Mai, MD Yamamoto, Mihoko, MD Aoki, Shigehisa, MD, PhD Toda, Shuji, MD, PhD |
| description | Summary Phosphaturic mesenchymal tumors of the mixed connective tissue type (PMT-MCTs) are rare neoplasms, most of which are benign and cause tumor-induced osteomalacia because of overproduction of a phosphaturic hormone, fibroblast growth factor 23 (FGF23). This entity may have been unrecognized or misdiagnosed as other mesenchymal tumors, such as giant cell tumor, hemangiopericytoma, and osteosarcoma. Ten percent of these tumors, without phosphaturia, were diagnosed only by their histologic features. We report here the first case of malignant PMT-MCT, nonphosphaturic variant, resulting in fatal multiple lung metastases. Chondromyxoid matrix with “grungy” calcification, multinucleated giant cell proliferation, and expression of FGF23 mRNA (reverse transcription–polymerase chain reaction) and fibroblast growth factor 23 protein (immunohistochemistry) were seen in the primary and recurrent tumors of the right foot. The lung metastases showed flocculent calcification and FGF23 protein expression as well as giant cell proliferation. This unique case highlights the need for careful histologic assessment of PMT-MCTs, especially the nonphosphaturic variant, and the need for recognition of its rare malignant behavior. |
| doi_str_mv | 10.1016/j.humpath.2013.04.027 |
| format | Article |
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This entity may have been unrecognized or misdiagnosed as other mesenchymal tumors, such as giant cell tumor, hemangiopericytoma, and osteosarcoma. Ten percent of these tumors, without phosphaturia, were diagnosed only by their histologic features. We report here the first case of malignant PMT-MCT, nonphosphaturic variant, resulting in fatal multiple lung metastases. Chondromyxoid matrix with “grungy” calcification, multinucleated giant cell proliferation, and expression of FGF23 mRNA (reverse transcription–polymerase chain reaction) and fibroblast growth factor 23 protein (immunohistochemistry) were seen in the primary and recurrent tumors of the right foot. The lung metastases showed flocculent calcification and FGF23 protein expression as well as giant cell proliferation. This unique case highlights the need for careful histologic assessment of PMT-MCTs, especially the nonphosphaturic variant, and the need for recognition of its rare malignant behavior.</description><identifier>ISSN: 0046-8177</identifier><identifier>EISSN: 1532-8392</identifier><identifier>DOI: 10.1016/j.humpath.2013.04.027</identifier><identifier>PMID: 23954138</identifier><language>eng</language><publisher>United States: Elsevier Inc</publisher><subject>Adipocytes ; Adult ; Calcinosis ; Cell growth ; Cell Proliferation ; Diagnosis, Differential ; Fatal Outcome ; Female ; FGF23 ; Fibroblast Growth Factors - metabolism ; Foot - pathology ; Giant cells ; Giant Cells - pathology ; Humans ; Hypophosphatemia, Familial ; Immunohistochemistry ; Lung - pathology ; Lung Neoplasms - pathology ; Lung Neoplasms - secondary ; Lung Neoplasms - surgery ; Mesenchymoma - metabolism ; Mesenchymoma - pathology ; Mesenchymoma - secondary ; Neoplasm Recurrence, Local - metabolism ; Neoplasm Recurrence, Local - pathology ; Neoplasms, Connective Tissue - metabolism ; Neoplasms, Connective Tissue - pathology ; Neoplasms, Connective Tissue - secondary ; Pathology ; Phosphaturic mesenchymal tumors of the mixed connective tissue type ; Pulmonary metastasis ; Reverse Transcriptase Polymerase Chain Reaction ; RNA, Messenger - genetics ; RNA, Neoplasm - genetics ; Soft Tissue Neoplasms - metabolism ; Soft Tissue Neoplasms - pathology ; Soft Tissue Neoplasms - surgery ; Thoracic surgery ; Tumors</subject><ispartof>Human pathology, 2013-11, Vol.44 (11), p.2614-2618</ispartof><rights>Elsevier Inc.</rights><rights>2013 Elsevier Inc.</rights><rights>Copyright © 2013 Elsevier Inc. All rights reserved.</rights><rights>Copyright Elsevier Limited Nov 2013</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c514t-75fa6e81b9fe515ac078c489d0775fae1d7de5919965d2d7d28a451f389f0cfb3</citedby><cites>FETCH-LOGICAL-c514t-75fa6e81b9fe515ac078c489d0775fae1d7de5919965d2d7d28a451f389f0cfb3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktohtml>$$Uhttps://dx.doi.org/10.1016/j.humpath.2013.04.027$$EHTML$$P50$$Gelsevier$$H</linktohtml><link.rule.ids>314,780,784,3550,27924,27925,45995</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/23954138$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Uchihashi, Kazuyoshi, MD, PhD</creatorcontrib><creatorcontrib>Nishijima-Matsunobu, Aki, MD, PhD</creatorcontrib><creatorcontrib>Matsuyama, Atsuji, MD, PhD</creatorcontrib><creatorcontrib>Yamasaki, Fumio, MD, PhD</creatorcontrib><creatorcontrib>Tanabe, Tsuyoshi, MD</creatorcontrib><creatorcontrib>Uemura, Tetsuji, MD, PhD</creatorcontrib><creatorcontrib>Aragane, Naoko, MD, PhD</creatorcontrib><creatorcontrib>Yakushiji, Mai, MD</creatorcontrib><creatorcontrib>Yamamoto, Mihoko, MD</creatorcontrib><creatorcontrib>Aoki, Shigehisa, MD, PhD</creatorcontrib><creatorcontrib>Toda, Shuji, MD, PhD</creatorcontrib><title>Phosphaturic mesenchymal tumor, nonphosphaturic variant, causing fatal pulmonary metastasis</title><title>Human pathology</title><addtitle>Hum Pathol</addtitle><description>Summary Phosphaturic mesenchymal tumors of the mixed connective tissue type (PMT-MCTs) are rare neoplasms, most of which are benign and cause tumor-induced osteomalacia because of overproduction of a phosphaturic hormone, fibroblast growth factor 23 (FGF23). This entity may have been unrecognized or misdiagnosed as other mesenchymal tumors, such as giant cell tumor, hemangiopericytoma, and osteosarcoma. Ten percent of these tumors, without phosphaturia, were diagnosed only by their histologic features. We report here the first case of malignant PMT-MCT, nonphosphaturic variant, resulting in fatal multiple lung metastases. Chondromyxoid matrix with “grungy” calcification, multinucleated giant cell proliferation, and expression of FGF23 mRNA (reverse transcription–polymerase chain reaction) and fibroblast growth factor 23 protein (immunohistochemistry) were seen in the primary and recurrent tumors of the right foot. The lung metastases showed flocculent calcification and FGF23 protein expression as well as giant cell proliferation. This unique case highlights the need for careful histologic assessment of PMT-MCTs, especially the nonphosphaturic variant, and the need for recognition of its rare malignant behavior.</description><subject>Adipocytes</subject><subject>Adult</subject><subject>Calcinosis</subject><subject>Cell growth</subject><subject>Cell Proliferation</subject><subject>Diagnosis, Differential</subject><subject>Fatal Outcome</subject><subject>Female</subject><subject>FGF23</subject><subject>Fibroblast Growth Factors - metabolism</subject><subject>Foot - pathology</subject><subject>Giant cells</subject><subject>Giant Cells - pathology</subject><subject>Humans</subject><subject>Hypophosphatemia, Familial</subject><subject>Immunohistochemistry</subject><subject>Lung - pathology</subject><subject>Lung Neoplasms - pathology</subject><subject>Lung Neoplasms - secondary</subject><subject>Lung Neoplasms - surgery</subject><subject>Mesenchymoma - metabolism</subject><subject>Mesenchymoma - pathology</subject><subject>Mesenchymoma - secondary</subject><subject>Neoplasm Recurrence, Local - metabolism</subject><subject>Neoplasm Recurrence, Local - pathology</subject><subject>Neoplasms, Connective Tissue - metabolism</subject><subject>Neoplasms, Connective Tissue - pathology</subject><subject>Neoplasms, Connective Tissue - secondary</subject><subject>Pathology</subject><subject>Phosphaturic mesenchymal tumors of the mixed connective tissue type</subject><subject>Pulmonary metastasis</subject><subject>Reverse Transcriptase Polymerase Chain Reaction</subject><subject>RNA, Messenger - genetics</subject><subject>RNA, Neoplasm - genetics</subject><subject>Soft Tissue Neoplasms - metabolism</subject><subject>Soft Tissue Neoplasms - pathology</subject><subject>Soft Tissue Neoplasms - surgery</subject><subject>Thoracic surgery</subject><subject>Tumors</subject><issn>0046-8177</issn><issn>1532-8392</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2013</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqFkl1rFDEUhoNY7Fr9CcqAN73ojOfkYyZzo5Riq1BQUEHwImQzGSfrfJlkCvvvzbCrlt4IgRDynDc5D4eQFwgFApavd0W3DLOOXUEBWQG8AFo9IhsUjOaS1fQx2QDwMpdYVafkaQg7AETBxRNySlktODK5Id8_dVOYOx0X70w22GBH0-0H3WdxGSZ_kY3TON9H7rR3eowXmdFLcOOPrNUx0fPSD9Oo_T5lRB3ScuEZOWl1H-zz435Gvl6_-3L1Pr_9ePPh6vI2NwJ5zCvR6tJK3NatFSi0gUoaLusGqvXKYlM1VtRY16VoaDpQqbnAlsm6BdNu2Rk5P-TOfvq12BDV4IKxfa9HOy1BIeeMIwhJE_rqAbqbFj-m361UWlBKTJQ4UMZPIXjbqtm7ITWnENRqX-3U0b5a7SvgKtlPdS-P6ct2sM3fqj-6E_D2ANik485Zr4JxybhtnLcmqmZy_33izYME07vRGd3_tHsb_nWjAlWgPq8jsE4AMgDK4Bv7DUy-rvg</recordid><startdate>20131101</startdate><enddate>20131101</enddate><creator>Uchihashi, Kazuyoshi, MD, PhD</creator><creator>Nishijima-Matsunobu, Aki, MD, PhD</creator><creator>Matsuyama, Atsuji, MD, PhD</creator><creator>Yamasaki, Fumio, MD, PhD</creator><creator>Tanabe, Tsuyoshi, MD</creator><creator>Uemura, Tetsuji, MD, PhD</creator><creator>Aragane, Naoko, MD, PhD</creator><creator>Yakushiji, Mai, MD</creator><creator>Yamamoto, Mihoko, MD</creator><creator>Aoki, Shigehisa, MD, PhD</creator><creator>Toda, Shuji, MD, PhD</creator><general>Elsevier Inc</general><general>Elsevier Limited</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>K9.</scope><scope>7X8</scope></search><sort><creationdate>20131101</creationdate><title>Phosphaturic mesenchymal tumor, nonphosphaturic variant, causing fatal pulmonary metastasis</title><author>Uchihashi, Kazuyoshi, MD, PhD ; Nishijima-Matsunobu, Aki, MD, PhD ; Matsuyama, Atsuji, MD, PhD ; Yamasaki, Fumio, MD, PhD ; Tanabe, Tsuyoshi, MD ; Uemura, Tetsuji, MD, PhD ; Aragane, Naoko, MD, PhD ; Yakushiji, Mai, MD ; Yamamoto, Mihoko, MD ; Aoki, Shigehisa, MD, PhD ; Toda, Shuji, MD, PhD</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c514t-75fa6e81b9fe515ac078c489d0775fae1d7de5919965d2d7d28a451f389f0cfb3</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2013</creationdate><topic>Adipocytes</topic><topic>Adult</topic><topic>Calcinosis</topic><topic>Cell growth</topic><topic>Cell Proliferation</topic><topic>Diagnosis, Differential</topic><topic>Fatal Outcome</topic><topic>Female</topic><topic>FGF23</topic><topic>Fibroblast Growth Factors - metabolism</topic><topic>Foot - pathology</topic><topic>Giant cells</topic><topic>Giant Cells - pathology</topic><topic>Humans</topic><topic>Hypophosphatemia, Familial</topic><topic>Immunohistochemistry</topic><topic>Lung - pathology</topic><topic>Lung Neoplasms - pathology</topic><topic>Lung Neoplasms - secondary</topic><topic>Lung Neoplasms - surgery</topic><topic>Mesenchymoma - metabolism</topic><topic>Mesenchymoma - pathology</topic><topic>Mesenchymoma - secondary</topic><topic>Neoplasm Recurrence, Local - metabolism</topic><topic>Neoplasm Recurrence, Local - pathology</topic><topic>Neoplasms, Connective Tissue - metabolism</topic><topic>Neoplasms, Connective Tissue - pathology</topic><topic>Neoplasms, Connective Tissue - secondary</topic><topic>Pathology</topic><topic>Phosphaturic mesenchymal tumors of the mixed connective tissue type</topic><topic>Pulmonary metastasis</topic><topic>Reverse Transcriptase Polymerase Chain Reaction</topic><topic>RNA, Messenger - genetics</topic><topic>RNA, Neoplasm - genetics</topic><topic>Soft Tissue Neoplasms - metabolism</topic><topic>Soft Tissue Neoplasms - pathology</topic><topic>Soft Tissue Neoplasms - surgery</topic><topic>Thoracic surgery</topic><topic>Tumors</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Uchihashi, Kazuyoshi, MD, PhD</creatorcontrib><creatorcontrib>Nishijima-Matsunobu, Aki, MD, PhD</creatorcontrib><creatorcontrib>Matsuyama, Atsuji, MD, PhD</creatorcontrib><creatorcontrib>Yamasaki, Fumio, MD, PhD</creatorcontrib><creatorcontrib>Tanabe, Tsuyoshi, MD</creatorcontrib><creatorcontrib>Uemura, Tetsuji, MD, PhD</creatorcontrib><creatorcontrib>Aragane, Naoko, MD, PhD</creatorcontrib><creatorcontrib>Yakushiji, Mai, MD</creatorcontrib><creatorcontrib>Yamamoto, Mihoko, MD</creatorcontrib><creatorcontrib>Aoki, Shigehisa, MD, PhD</creatorcontrib><creatorcontrib>Toda, Shuji, MD, PhD</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 Health & Medical Complete (Alumni)</collection><collection>MEDLINE - Academic</collection><jtitle>Human pathology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Uchihashi, Kazuyoshi, MD, PhD</au><au>Nishijima-Matsunobu, Aki, MD, PhD</au><au>Matsuyama, Atsuji, MD, PhD</au><au>Yamasaki, Fumio, MD, PhD</au><au>Tanabe, Tsuyoshi, MD</au><au>Uemura, Tetsuji, MD, PhD</au><au>Aragane, Naoko, MD, PhD</au><au>Yakushiji, Mai, MD</au><au>Yamamoto, Mihoko, MD</au><au>Aoki, Shigehisa, MD, PhD</au><au>Toda, Shuji, MD, PhD</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Phosphaturic mesenchymal tumor, nonphosphaturic variant, causing fatal pulmonary metastasis</atitle><jtitle>Human pathology</jtitle><addtitle>Hum Pathol</addtitle><date>2013-11-01</date><risdate>2013</risdate><volume>44</volume><issue>11</issue><spage>2614</spage><epage>2618</epage><pages>2614-2618</pages><issn>0046-8177</issn><eissn>1532-8392</eissn><abstract>Summary Phosphaturic mesenchymal tumors of the mixed connective tissue type (PMT-MCTs) are rare neoplasms, most of which are benign and cause tumor-induced osteomalacia because of overproduction of a phosphaturic hormone, fibroblast growth factor 23 (FGF23). This entity may have been unrecognized or misdiagnosed as other mesenchymal tumors, such as giant cell tumor, hemangiopericytoma, and osteosarcoma. Ten percent of these tumors, without phosphaturia, were diagnosed only by their histologic features. We report here the first case of malignant PMT-MCT, nonphosphaturic variant, resulting in fatal multiple lung metastases. Chondromyxoid matrix with “grungy” calcification, multinucleated giant cell proliferation, and expression of FGF23 mRNA (reverse transcription–polymerase chain reaction) and fibroblast growth factor 23 protein (immunohistochemistry) were seen in the primary and recurrent tumors of the right foot. The lung metastases showed flocculent calcification and FGF23 protein expression as well as giant cell proliferation. This unique case highlights the need for careful histologic assessment of PMT-MCTs, especially the nonphosphaturic variant, and the need for recognition of its rare malignant behavior.</abstract><cop>United States</cop><pub>Elsevier Inc</pub><pmid>23954138</pmid><doi>10.1016/j.humpath.2013.04.027</doi><tpages>5</tpages></addata></record> |
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| subjects | Adipocytes Adult Calcinosis Cell growth Cell Proliferation Diagnosis, Differential Fatal Outcome Female FGF23 Fibroblast Growth Factors - metabolism Foot - pathology Giant cells Giant Cells - pathology Humans Hypophosphatemia, Familial Immunohistochemistry Lung - pathology Lung Neoplasms - pathology Lung Neoplasms - secondary Lung Neoplasms - surgery Mesenchymoma - metabolism Mesenchymoma - pathology Mesenchymoma - secondary Neoplasm Recurrence, Local - metabolism Neoplasm Recurrence, Local - pathology Neoplasms, Connective Tissue - metabolism Neoplasms, Connective Tissue - pathology Neoplasms, Connective Tissue - secondary Pathology Phosphaturic mesenchymal tumors of the mixed connective tissue type Pulmonary metastasis Reverse Transcriptase Polymerase Chain Reaction RNA, Messenger - genetics RNA, Neoplasm - genetics Soft Tissue Neoplasms - metabolism Soft Tissue Neoplasms - pathology Soft Tissue Neoplasms - surgery Thoracic surgery Tumors |
| title | Phosphaturic mesenchymal tumor, nonphosphaturic variant, causing fatal pulmonary metastasis |
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