Desmoplastic Small Round Cell Tumor: Report of 2 Cases Treated with Chemotherapy Alone or in Combination with Bevacizumab
Identifieur interne : 001C32 ( Pmc/Checkpoint ); précédent : 001C31; suivant : 001C33Desmoplastic Small Round Cell Tumor: Report of 2 Cases Treated with Chemotherapy Alone or in Combination with Bevacizumab
Auteurs : Rogério Agenor De Araújo [Brésil] ; Breno Jeha AraújoSource :
- Case Reports in Oncology [ 1662-6575 ] ; 2014.
Abstract
Here, we present 2 case reports of patients with desmoplastic small round cell tumor (DSRCT), a very rare and aggressive mesenchymal cancer, and we discuss 2therapeutic options for this sarcoma. This report focuses on men aged 22 and 37 years, respectively. The first patient presented with an abdominopelvic mass which was not suitable for surgery. He underwent chemotherapy (adriblastina and cisplatin) with a brief partial remission and survival time of 13 months. The second patient presented with an abdominal mass and underwent partial resection. He received chemotherapy and bevacizumab, resulting in a partial remission and a survival time of 34 months. The extent of surgery and monoclonal antibody use probably had a positive impact on survival. It is necessary to include specific targeted therapies in an attempt to improve survival. Surprisingly, positron emission tomography was not effective in restaging of the second patient, emphasizing the importance of computed tomography or magnetic resonance in DSRCT.
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DOI: 10.1159/000359997
PubMed: 24707256
PubMed Central: 3975756
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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Desmoplastic Small Round Cell Tumor: Report of 2 Cases Treated with Chemotherapy Alone or in Combination with Bevacizumab</title><author><name sortKey="De Araujo, Rogerio Agenor" sort="De Araujo, Rogerio Agenor" uniqKey="De Araujo R" first="Rogério Agenor" last="De Araújo">Rogério Agenor De Araújo</name><affiliation wicri:level="1"><nlm:aff id="aff1">Department of Oncology, Federal University of Uberlândia, Uberlândia, Brazil</nlm:aff><country xml:lang="fr">Brésil</country><wicri:regionArea>Department of Oncology, Federal University of Uberlândia, Uberlândia</wicri:regionArea><wicri:noRegion>Uberlândia</wicri:noRegion></affiliation></author><author><name sortKey="Araujo, Breno Jeha" sort="Araujo, Breno Jeha" uniqKey="Araujo B" first="Breno Jeha" last="Araújo">Breno Jeha Araújo</name><affiliation><nlm:aff>NONE</nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">24707256</idno><idno type="pmc">3975756</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3975756</idno><idno type="RBID">PMC:3975756</idno><idno type="doi">10.1159/000359997</idno><date when="2014">2014</date><idno type="wicri:Area/Pmc/Corpus">002146</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">002146</idno><idno type="wicri:Area/Pmc/Curation">002145</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Curation">002145</idno><idno type="wicri:Area/Pmc/Checkpoint">001C32</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Checkpoint">001C32</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Desmoplastic Small Round Cell Tumor: Report of 2 Cases Treated with Chemotherapy Alone or in Combination with Bevacizumab</title><author><name sortKey="De Araujo, Rogerio Agenor" sort="De Araujo, Rogerio Agenor" uniqKey="De Araujo R" first="Rogério Agenor" last="De Araújo">Rogério Agenor De Araújo</name><affiliation wicri:level="1"><nlm:aff id="aff1">Department of Oncology, Federal University of Uberlândia, Uberlândia, Brazil</nlm:aff><country xml:lang="fr">Brésil</country><wicri:regionArea>Department of Oncology, Federal University of Uberlândia, Uberlândia</wicri:regionArea><wicri:noRegion>Uberlândia</wicri:noRegion></affiliation></author><author><name sortKey="Araujo, Breno Jeha" sort="Araujo, Breno Jeha" uniqKey="Araujo B" first="Breno Jeha" last="Araújo">Breno Jeha Araújo</name><affiliation><nlm:aff>NONE</nlm:aff></affiliation></author></analytic><series><title level="j">Case Reports in Oncology</title><idno type="eISSN">1662-6575</idno><imprint><date when="2014">2014</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Here, we present 2 case reports of patients with desmoplastic small round cell tumor (DSRCT), a very rare and aggressive mesenchymal cancer, and we discuss 2therapeutic options for this sarcoma. This report focuses on men aged 22 and 37 years, respectively. The first patient presented with an abdominopelvic mass which was not suitable for surgery. He underwent chemotherapy (adriblastina and cisplatin) with a brief partial remission and survival time of 13 months. The second patient presented with an abdominal mass and underwent partial resection. He received chemotherapy and bevacizumab, resulting in a partial remission and a survival time of 34 months. The extent of surgery and monoclonal antibody use probably had a positive impact on survival. It is necessary to include specific targeted therapies in an attempt to improve survival. Surprisingly, positron emission tomography was not effective in restaging of the second patient, emphasizing the importance of computed tomography or magnetic resonance in DSRCT.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Koniari, K" uniqKey="Koniari K">K Koniari</name></author><author><name sortKey="Mahera, H" uniqKey="Mahera H">H Mahera</name></author><author><name sortKey="Nikolaou, M" uniqKey="Nikolaou M">M Nikolaou</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Dufresne, A" uniqKey="Dufresne A">A Dufresne</name></author><author><name sortKey="Cassier, P" uniqKey="Cassier P">P Cassier</name></author><author><name sortKey="Couraud, L" uniqKey="Couraud L">L Couraud</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Perna, Mj" uniqKey="Perna M">MJ Perna</name></author><author><name sortKey="Streck, Cj" uniqKey="Streck C">CJ Streck</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Gerald, Wl" uniqKey="Gerald W">WL Gerald</name></author><author><name sortKey="Rosai, J" uniqKey="Rosai J">J Rosai</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey=" Rdo Ez, Ng" uniqKey=" Rdo Ez N">NG Órdoñez</name></author><author><name sortKey="El Naggar, Ak" uniqKey="El Naggar A">AK El-Naggar</name></author><author><name sortKey="Ro, Jy" uniqKey="Ro J">JY Ro</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Kretschmar, Cs" uniqKey="Kretschmar C">CS Kretschmar</name></author><author><name sortKey="Colbach, C" uniqKey="Colbach C">C Colbach</name></author><author><name sortKey="Bhan, I" uniqKey="Bhan I">I Bhan</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Gerald, Wl" uniqKey="Gerald W">WL Gerald</name></author><author><name sortKey="Ladanyi, M" uniqKey="Ladanyi M">M Ladanyi</name></author><author><name sortKey="Alava, E" uniqKey="Alava E">E Alava</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Ogata, Dc" uniqKey="Ogata D">DC Ogata</name></author><author><name sortKey="Tatsugui, Jt" uniqKey="Tatsugui J">JT Tatsugui</name></author><author><name sortKey="Machuca, Tn" uniqKey="Machuca T">TN Machuca</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Cao, L" uniqKey="Cao L">L Cao</name></author><author><name sortKey="Ni, J" uniqKey="Ni J">J Ni</name></author><author><name sortKey="Que, R" uniqKey="Que R">R Que</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Philippe Chomette, P" uniqKey="Philippe Chomette P">P Philippe-Chomette</name></author><author><name sortKey="Kabbara, N" uniqKey="Kabbara N">N Kabbara</name></author><author><name sortKey="Andre, N" uniqKey="Andre N">N Andre</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Torres, Us" uniqKey="Torres U">US Torres</name></author><author><name sortKey="Ribeiro, Mca" uniqKey="Ribeiro M">MCA Ribeiro</name></author><author><name sortKey="Souza, As" uniqKey="Souza A">AS Souza</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Kushner, Bh" uniqKey="Kushner B">BH Kushner</name></author><author><name sortKey="Laquaglia, Mp" uniqKey="Laquaglia M">MP LaQuaglia</name></author><author><name sortKey="Wollner, N" uniqKey="Wollner N">N Wollner</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Gil, A" uniqKey="Gil A">A Gil</name></author><author><name sortKey="Portilla, Ag" uniqKey="Portilla A">AG Portilla</name></author><author><name sortKey="Brun, Ea" uniqKey="Brun E">EA Brun</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Mrabti, H" uniqKey="Mrabti H">H Mrabti</name></author><author><name sortKey="Kaikani, W" uniqKey="Kaikani W">W Kaikani</name></author><author><name sortKey="Ahbeddou, N" uniqKey="Ahbeddou N">N Ahbeddou</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Westphalen, Aca" uniqKey="Westphalen A">ACA Westphalen</name></author><author><name sortKey="Ferreira, Jhp" uniqKey="Ferreira J">JHP Ferreira</name></author><author><name sortKey="Daudt, Aw" uniqKey="Daudt A">AW Daudt</name></author></analytic></biblStruct></listBibl></div1></back></TEI><pmc article-type="case-report"><pmc-dir>properties open_access</pmc-dir>
<front><journal-meta><journal-id journal-id-type="nlm-ta">Case Rep Oncol</journal-id><journal-id journal-id-type="iso-abbrev">Case Rep Oncol</journal-id><journal-id journal-id-type="publisher-id">CRO</journal-id><journal-title-group><journal-title>Case Reports in Oncology</journal-title></journal-title-group><issn pub-type="epub">1662-6575</issn><publisher><publisher-name>S. Karger AG</publisher-name><publisher-loc>Allschwilerstrasse 10, P.O. Box · Postfach · Case postale, CH–4009, Basel, Switzerland · Schweiz · Suisse, Phone: +41 61 306 11 11, Fax: +41 61 306 12 34, karger@karger.ch</publisher-loc></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">24707256</article-id><article-id pub-id-type="pmc">3975756</article-id><article-id pub-id-type="doi">10.1159/000359997</article-id><article-id pub-id-type="publisher-id">cro-0007-0102</article-id><article-categories><subj-group subj-group-type="heading"><subject>Published online: February, 2014</subject></subj-group></article-categories><title-group><article-title>Desmoplastic Small Round Cell Tumor: Report of 2 Cases Treated with Chemotherapy Alone or in Combination with Bevacizumab</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>de Araújo</surname><given-names>Rogério Agenor</given-names></name><xref ref-type="aff" rid="aff1"><sup>a</sup></xref><xref ref-type="corresp" rid="cor1">*</xref></contrib><contrib contrib-type="author"><name><surname>Araújo</surname><given-names>Breno Jeha</given-names></name><xref ref-type="aff" rid="aff2"><sup>b</sup></xref></contrib></contrib-group><aff id="aff1"><sup>a</sup>Department of Oncology, Federal University of Uberlândia, Uberlândia, Brazil</aff><aff id="aff2"><sup>2</sup>School of Medicine, Federal University of Uberlândia, Uberlândia, Brazil</aff><author-notes><corresp id="cor1">*Rogério Agenor de Araújo, Department of Oncology, Universidade Federal de Uberlândia, Campus Umuarama, Bloco 4A, Uberlândia, MG 38400-000 (Brazil), E-Mail <email>rogeriodearaujo@gmail.com</email></corresp></author-notes><pub-date pub-type="collection"><season>Jan-Apr</season><year>2014</year></pub-date><pub-date pub-type="epub"><day>15</day><month>2</month><year>2014</year></pub-date><pub-date pub-type="pmc-release"><day>15</day><month>2</month><year>2014</year></pub-date><pmc-comment> PMC Release delay is 0 months and 0 days and was based on the
<volume>7</volume><issue>1</issue><fpage>102</fpage><lpage>108</lpage><permissions><copyright-statement>Copyright © 2014 by S. Karger AG, Basel</copyright-statement><copyright-year>2014</copyright-year><license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc/3.0/"><license-p>This is an Open Access article licensed under the terms of the Creative Commons Attribution-NonCommercial 3.0 Unported license (CC BY-NC) (www.karger.com/OA-license), applicable to the online version of the article only. Users may download, print and share this work on the Internet for noncommercial purposes only, provided the original work is properly cited, and a link to the original work on http://www.karger.com and the terms of this license are included in any shared versions.</license-p></license></permissions><abstract><p>Here, we present 2 case reports of patients with desmoplastic small round cell tumor (DSRCT), a very rare and aggressive mesenchymal cancer, and we discuss 2therapeutic options for this sarcoma. This report focuses on men aged 22 and 37 years, respectively. The first patient presented with an abdominopelvic mass which was not suitable for surgery. He underwent chemotherapy (adriblastina and cisplatin) with a brief partial remission and survival time of 13 months. The second patient presented with an abdominal mass and underwent partial resection. He received chemotherapy and bevacizumab, resulting in a partial remission and a survival time of 34 months. The extent of surgery and monoclonal antibody use probably had a positive impact on survival. It is necessary to include specific targeted therapies in an attempt to improve survival. Surprisingly, positron emission tomography was not effective in restaging of the second patient, emphasizing the importance of computed tomography or magnetic resonance in DSRCT.</p></abstract><kwd-group><title>Key words</title><kwd>Desmoplastic small round cell tumor</kwd><kwd>Chemotherapy</kwd><kwd>Abdominal tumor</kwd><kwd>Bevacizumab</kwd></kwd-group><counts><fig-count count="3"></fig-count><table-count count="1"></table-count><ref-count count="15"></ref-count><page-count count="7"></page-count></counts></article-meta></front><floats-group><fig id="F1" orientation="portrait" position="float"><label>Fig. 1</label><caption><p>Case 1: CT scan showing a retrovesical tumor.</p></caption><graphic xlink:href="cro-0007-0102-g01"></graphic></fig><fig id="F2" orientation="portrait" position="float"><label>Fig. 2</label><caption><p>Case 2: CT scans showing hilum and hepatic parenchyma infiltration (PET without any uptake).</p></caption><graphic xlink:href="cro-0007-0102-g02"></graphic></fig><fig id="F3" orientation="portrait" position="float"><label>Fig. 3</label><caption><p>Case 3: physiological distribution of FDG shown on PET without any uptake.</p></caption><graphic xlink:href="cro-0007-0102-g03"></graphic></fig><table-wrap id="T1" orientation="portrait" position="float"><label>Table 1</label><caption><p>IH studies compatible with DSRCT</p></caption><table frame="hsides" rules="groups"><thead><tr valign="top"><th align="left" rowspan="1" colspan="1">Antibody used</th><th align="left" rowspan="1" colspan="1">Case 1</th><th align="left" rowspan="1" colspan="1">Case 2</th></tr></thead><tbody><tr valign="top"><td align="left" rowspan="1" colspan="1">Anti-cytokeratins</td><td align="left" rowspan="1" colspan="1">Positive</td><td align="left" rowspan="1" colspan="1">Positive</td></tr><tr valign="top"><td align="left" rowspan="1" colspan="1">Anti-EMA/E29</td><td align="left" rowspan="1" colspan="1">Positive</td><td align="left" rowspan="1" colspan="1">–</td></tr><tr valign="top"><td align="left" rowspan="1" colspan="1">Anti-desmin/D33</td><td align="left" rowspan="1" colspan="1">Positive</td><td align="left" rowspan="1" colspan="1">Positive</td></tr><tr valign="top"><td align="left" rowspan="1" colspan="1">Anti-NSE</td><td align="left" rowspan="1" colspan="1">Positive</td><td align="left" rowspan="1" colspan="1">–</td></tr><tr valign="top"><td align="left" rowspan="1" colspan="1">Anti-vimentin</td><td align="left" rowspan="1" colspan="1">Positive</td><td align="left" rowspan="1" colspan="1">–</td></tr><tr valign="top"><td align="left" rowspan="1" colspan="1">Anti-gene MIC2 (CD99)</td><td align="left" rowspan="1" colspan="1">Positive</td><td align="left" rowspan="1" colspan="1">Negative</td></tr><tr valign="top"><td align="left" rowspan="1" colspan="1">Anti-chromogranin</td><td align="left" rowspan="1" colspan="1">Negative</td><td align="left" rowspan="1" colspan="1">Negative</td></tr><tr valign="top"><td align="left" rowspan="1" colspan="1">Anti-LCA</td><td align="left" rowspan="1" colspan="1">Negative</td><td align="left" rowspan="1" colspan="1">–</td></tr><tr valign="top"><td align="left" rowspan="1" colspan="1">Anti-protein S100</td><td align="left" rowspan="1" colspan="1">Focal positivity</td><td align="left" rowspan="1" colspan="1">–</td></tr><tr valign="top"><td align="left" rowspan="1" colspan="1">Anti-synaptophysin</td><td align="left" rowspan="1" colspan="1">Negative</td><td align="left" rowspan="1" colspan="1">Negative</td></tr><tr valign="top"><td align="left" rowspan="1" colspan="1">Cytokeratins 8 and 18</td><td align="left" rowspan="1" colspan="1">–</td><td align="left" rowspan="1" colspan="1">Positive</td></tr><tr valign="top"><td align="left" rowspan="1" colspan="1">Enolase</td><td align="left" rowspan="1" colspan="1">–</td><td align="left" rowspan="1" colspan="1">Positive</td></tr><tr valign="top"><td align="left" rowspan="1" colspan="1">Cytokeratin 20</td><td align="left" rowspan="1" colspan="1">–</td><td align="left" rowspan="1" colspan="1">Negative</td></tr><tr valign="top"><td align="left" rowspan="1" colspan="1">CDX-2</td><td align="left" rowspan="1" colspan="1">–</td><td align="left" rowspan="1" colspan="1">Negative</td></tr></tbody></table></table-wrap></floats-group></pmc><affiliations><list><country><li>Brésil</li></country></list><tree><noCountry><name sortKey="Araujo, Breno Jeha" sort="Araujo, Breno Jeha" uniqKey="Araujo B" first="Breno Jeha" last="Araújo">Breno Jeha Araújo</name></noCountry><country name="Brésil"><noRegion><name sortKey="De Araujo, Rogerio Agenor" sort="De Araujo, Rogerio Agenor" uniqKey="De Araujo R" first="Rogério Agenor" last="De Araújo">Rogério Agenor De Araújo</name></noRegion></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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