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Cytotoxic and targeted therapy for treatment of pseudomyogenic hemangioendothelioma
Pseudomyogenic hemangioendothelioma (PMH) is a recently described, indolent vascular tumor that usually presents in the distal extremities. PMH typically has a multi-focal presentation and can involve several tissue planes including the dermis, subcutis, muscle, and bone. This soft tissue tumor pred...
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Published in: | Clinical sarcoma research 2015-10, Vol.5 (1), p.22, Article 22 |
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description | Pseudomyogenic hemangioendothelioma (PMH) is a recently described, indolent vascular tumor that usually presents in the distal extremities. PMH typically has a multi-focal presentation and can involve several tissue planes including the dermis, subcutis, muscle, and bone. This soft tissue tumor predominantly affects men between 20 and 50 years of age. PMH tumors typically are resected but frequently recur locally; thus, more efficacious treatment options are needed. Herein, we report two cases of patients with PMH who were treated with systemic therapy. To the best of our knowledge, our report is the first to describe a response of PMH either to gemcitabine/taxane cytotoxic chemotherapy or to a mammalian target of rapamycin inhibitor. In the first case, a 45-year-old man with PMH of the right ilium was treated with gemcitabine plus docetaxel. Although chemotherapy was ultimately halted owing to gemcitabine-induced pulmonary toxicity, positron emission tomography-computer tomography scans taken after three cycles of gemcitabine plus docetaxel illustrated a noticeable response to the regimen. In the second case, a 22-year-old man with PMH of the right distal femur and metastases in the left ilium showed no response to gemcitabine plus docetaxel therapy, but underwent surgical resection after cisplatin and doxorubicin resulted in stable disease. DNA sequencing of his tumor revealed the presence of a tuberous sclerosis 1 (TSC1) mutation, so daily everolimus, which inhibits mammalian target of rapamycin, was started. Two months after beginning everolimus, the patient underwent magnetic resonance imaging of the pelvis, which revealed mild shrinkage of PMH metastases in the left iliac bone. Despite the apparent heterogeneity of response to gemcitabine/taxane chemotherapy in our two patients, these two cases indicate that gemcitabine/taxane and mammalian target of rapamycin inhibitor may serve as systemic treatment options for PMH and warrant further investigation. |
doi_str_mv | 10.1186/s13569-015-0037-8 |
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PMH typically has a multi-focal presentation and can involve several tissue planes including the dermis, subcutis, muscle, and bone. This soft tissue tumor predominantly affects men between 20 and 50 years of age. PMH tumors typically are resected but frequently recur locally; thus, more efficacious treatment options are needed. Herein, we report two cases of patients with PMH who were treated with systemic therapy. To the best of our knowledge, our report is the first to describe a response of PMH either to gemcitabine/taxane cytotoxic chemotherapy or to a mammalian target of rapamycin inhibitor. In the first case, a 45-year-old man with PMH of the right ilium was treated with gemcitabine plus docetaxel. Although chemotherapy was ultimately halted owing to gemcitabine-induced pulmonary toxicity, positron emission tomography-computer tomography scans taken after three cycles of gemcitabine plus docetaxel illustrated a noticeable response to the regimen. In the second case, a 22-year-old man with PMH of the right distal femur and metastases in the left ilium showed no response to gemcitabine plus docetaxel therapy, but underwent surgical resection after cisplatin and doxorubicin resulted in stable disease. DNA sequencing of his tumor revealed the presence of a tuberous sclerosis 1 (TSC1) mutation, so daily everolimus, which inhibits mammalian target of rapamycin, was started. Two months after beginning everolimus, the patient underwent magnetic resonance imaging of the pelvis, which revealed mild shrinkage of PMH metastases in the left iliac bone. Despite the apparent heterogeneity of response to gemcitabine/taxane chemotherapy in our two patients, these two cases indicate that gemcitabine/taxane and mammalian target of rapamycin inhibitor may serve as systemic treatment options for PMH and warrant further investigation.</description><identifier>ISSN: 2045-3329</identifier><identifier>EISSN: 2045-3329</identifier><identifier>DOI: 10.1186/s13569-015-0037-8</identifier><identifier>PMID: 26500758</identifier><language>eng</language><publisher>England: BioMed Central</publisher><subject>Case Report</subject><ispartof>Clinical sarcoma research, 2015-10, Vol.5 (1), p.22, Article 22</ispartof><rights>Copyright BioMed Central 2015</rights><rights>Joseph et al. 2015</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c457t-31bc523b8244e5172da30ee012e73fefe8d7697d47a4ad3fb8760833d8009733</citedby><cites>FETCH-LOGICAL-c457t-31bc523b8244e5172da30ee012e73fefe8d7697d47a4ad3fb8760833d8009733</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC4615364/pdf/$$EPDF$$P50$$Gpubmedcentral$$Hfree_for_read</linktopdf><linktohtml>$$Uhttps://www.proquest.com/docview/1772124039?pq-origsite=primo$$EHTML$$P50$$Gproquest$$Hfree_for_read</linktohtml><link.rule.ids>230,315,733,786,790,891,25783,27957,27958,37047,44625,53827,53829</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/26500758$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Joseph, Jason</creatorcontrib><creatorcontrib>Wang, Wei-Lien</creatorcontrib><creatorcontrib>Patnana, Madhavi</creatorcontrib><creatorcontrib>Ramesh, Naveen</creatorcontrib><creatorcontrib>Benjamin, Robert</creatorcontrib><creatorcontrib>Patel, Shreyaskumar</creatorcontrib><creatorcontrib>Ravi, Vinod</creatorcontrib><title>Cytotoxic and targeted therapy for treatment of pseudomyogenic hemangioendothelioma</title><title>Clinical sarcoma research</title><addtitle>Clin Sarcoma Res</addtitle><description>Pseudomyogenic hemangioendothelioma (PMH) is a recently described, indolent vascular tumor that usually presents in the distal extremities. PMH typically has a multi-focal presentation and can involve several tissue planes including the dermis, subcutis, muscle, and bone. This soft tissue tumor predominantly affects men between 20 and 50 years of age. PMH tumors typically are resected but frequently recur locally; thus, more efficacious treatment options are needed. Herein, we report two cases of patients with PMH who were treated with systemic therapy. To the best of our knowledge, our report is the first to describe a response of PMH either to gemcitabine/taxane cytotoxic chemotherapy or to a mammalian target of rapamycin inhibitor. In the first case, a 45-year-old man with PMH of the right ilium was treated with gemcitabine plus docetaxel. Although chemotherapy was ultimately halted owing to gemcitabine-induced pulmonary toxicity, positron emission tomography-computer tomography scans taken after three cycles of gemcitabine plus docetaxel illustrated a noticeable response to the regimen. In the second case, a 22-year-old man with PMH of the right distal femur and metastases in the left ilium showed no response to gemcitabine plus docetaxel therapy, but underwent surgical resection after cisplatin and doxorubicin resulted in stable disease. DNA sequencing of his tumor revealed the presence of a tuberous sclerosis 1 (TSC1) mutation, so daily everolimus, which inhibits mammalian target of rapamycin, was started. Two months after beginning everolimus, the patient underwent magnetic resonance imaging of the pelvis, which revealed mild shrinkage of PMH metastases in the left iliac bone. Despite the apparent heterogeneity of response to gemcitabine/taxane chemotherapy in our two patients, these two cases indicate that gemcitabine/taxane and mammalian target of rapamycin inhibitor may serve as systemic treatment options for PMH and warrant further investigation.</description><subject>Case Report</subject><issn>2045-3329</issn><issn>2045-3329</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2015</creationdate><recordtype>article</recordtype><sourceid>PIMPY</sourceid><recordid>eNpVUU1LAzEQDaLYUvsDvMiC59XJ1yZ7EaT4BQUP9h7SzWy7pbup2VTsvzelWupcZmDee_OYR8g1hTtKdXHfUy6LMgcqcwCucn1GhgyEzDln5fnJPCDjvl9BqgKkAnlJBqyQAErqIfmY7KKP_rupMtu5LNqwwIhpWGKwm11W-5DFgDa22MXM19mmx63z7c4vsEukJba2WzQeO-cTZ9341l6Ri9quexz_9hGZPT_NJq_59P3lbfI4zSshVcw5nVeS8blmQqCkijnLAREoQ8VrrFE7VZTKCWWFdbyea1WA5txpgFJxPiIPB9nNdt6iq5LBYNdmE5rWhp3xtjH_N12zNAv_ZURBJS9EErj9FQj-c4t9NCu_DV2ybKhSjDIBvEwoekBVwfd9wPp4gYLZJ2EOSZiUhNknYXTi3JxaOzL-_s5_AMYzhcI</recordid><startdate>20151019</startdate><enddate>20151019</enddate><creator>Joseph, Jason</creator><creator>Wang, Wei-Lien</creator><creator>Patnana, Madhavi</creator><creator>Ramesh, Naveen</creator><creator>Benjamin, Robert</creator><creator>Patel, Shreyaskumar</creator><creator>Ravi, Vinod</creator><general>BioMed Central</general><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7X7</scope><scope>7XB</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AZQEC</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>DWQXO</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>K9.</scope><scope>M0S</scope><scope>PIMPY</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>5PM</scope></search><sort><creationdate>20151019</creationdate><title>Cytotoxic and targeted therapy for treatment of pseudomyogenic hemangioendothelioma</title><author>Joseph, Jason ; Wang, Wei-Lien ; Patnana, Madhavi ; Ramesh, Naveen ; Benjamin, Robert ; Patel, Shreyaskumar ; Ravi, Vinod</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c457t-31bc523b8244e5172da30ee012e73fefe8d7697d47a4ad3fb8760833d8009733</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2015</creationdate><topic>Case Report</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Joseph, Jason</creatorcontrib><creatorcontrib>Wang, Wei-Lien</creatorcontrib><creatorcontrib>Patnana, Madhavi</creatorcontrib><creatorcontrib>Ramesh, Naveen</creatorcontrib><creatorcontrib>Benjamin, Robert</creatorcontrib><creatorcontrib>Patel, Shreyaskumar</creatorcontrib><creatorcontrib>Ravi, Vinod</creatorcontrib><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</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 Essentials</collection><collection>ProQuest Databases</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central Korea</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Publicly Available Content Database</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>PubMed Central (Full Participant titles)</collection><jtitle>Clinical sarcoma research</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Joseph, Jason</au><au>Wang, Wei-Lien</au><au>Patnana, Madhavi</au><au>Ramesh, Naveen</au><au>Benjamin, Robert</au><au>Patel, Shreyaskumar</au><au>Ravi, Vinod</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Cytotoxic and targeted therapy for treatment of pseudomyogenic hemangioendothelioma</atitle><jtitle>Clinical sarcoma research</jtitle><addtitle>Clin Sarcoma Res</addtitle><date>2015-10-19</date><risdate>2015</risdate><volume>5</volume><issue>1</issue><spage>22</spage><pages>22-</pages><artnum>22</artnum><issn>2045-3329</issn><eissn>2045-3329</eissn><abstract>Pseudomyogenic hemangioendothelioma (PMH) is a recently described, indolent vascular tumor that usually presents in the distal extremities. PMH typically has a multi-focal presentation and can involve several tissue planes including the dermis, subcutis, muscle, and bone. This soft tissue tumor predominantly affects men between 20 and 50 years of age. PMH tumors typically are resected but frequently recur locally; thus, more efficacious treatment options are needed. Herein, we report two cases of patients with PMH who were treated with systemic therapy. To the best of our knowledge, our report is the first to describe a response of PMH either to gemcitabine/taxane cytotoxic chemotherapy or to a mammalian target of rapamycin inhibitor. In the first case, a 45-year-old man with PMH of the right ilium was treated with gemcitabine plus docetaxel. Although chemotherapy was ultimately halted owing to gemcitabine-induced pulmonary toxicity, positron emission tomography-computer tomography scans taken after three cycles of gemcitabine plus docetaxel illustrated a noticeable response to the regimen. In the second case, a 22-year-old man with PMH of the right distal femur and metastases in the left ilium showed no response to gemcitabine plus docetaxel therapy, but underwent surgical resection after cisplatin and doxorubicin resulted in stable disease. DNA sequencing of his tumor revealed the presence of a tuberous sclerosis 1 (TSC1) mutation, so daily everolimus, which inhibits mammalian target of rapamycin, was started. Two months after beginning everolimus, the patient underwent magnetic resonance imaging of the pelvis, which revealed mild shrinkage of PMH metastases in the left iliac bone. 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title | Cytotoxic and targeted therapy for treatment of pseudomyogenic hemangioendothelioma |
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