Cutaneous angiosarcoma of the buttock complicated by severe thrombocytopenia: A case report
Identifieur interne : 004212 ( Pmc/Corpus ); précédent : 004211; suivant : 004213Cutaneous angiosarcoma of the buttock complicated by severe thrombocytopenia: A case report
Auteurs : Kaoru Nagao ; Kayo Suzuki ; Taketoshi Yasuda ; Takeshi Hori ; Jun Hachinoda ; Masahiko Kanamori ; Tomoatsu KimuraSource :
- Molecular and Clinical Oncology [ 2049-9450 ] ; 2013.
Abstract
Angiosarcoma (AS) is an aggressive, malignant endothelial cell tumor of vascular or lymphatic origin, the presentation and clinical behavior of which may vary according to its location. This is the case report of a 56-year-old woman with cutaneous angiosarcoma (CAS) of the buttock complicated by severe thrombocytopenia. A review of the literature revealed that only nine cases of CAS with thrombocytopenia have been previously reported. The prognosis of CAS complicated by thrombocytopenia is poor, even after treatment with combined chemotherapy and radiotherapy (RT). The composite karyotype was 46,XX,t(12;20)(p13;p11.2)[3]/47,X,add(X)(q13),del(6)(q?),add(12)(p13),−21,+2mar[2]/45,XX,der(1)add(1)(p36.3)del(1)(q41),−20[1]/46,XX[13]. Only 13 cytogenetic cases of AS, including the present case, have been reported in the English literature thus far. In this case report, the clinical presentation and cytogenetic findings are described and the relevant literature on AS is reviewed.
Url:
DOI: 10.3892/mco.2013.141
PubMed: 24649268
PubMed Central: 3915322
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PMC:3915322Le document en format XML
<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Cutaneous angiosarcoma of the buttock complicated by severe thrombocytopenia: A case report</title><author><name sortKey="Nagao, Kaoru" sort="Nagao, Kaoru" uniqKey="Nagao K" first="Kaoru" last="Nagao">Kaoru Nagao</name><affiliation><nlm:aff id="af1-mco-01-05-0903">Department of Orthopaedic Surgery, Faculty of Medicine, University of Toyama, Toyama, Toyama 930-0194, Japan</nlm:aff></affiliation></author><author><name sortKey="Suzuki, Kayo" sort="Suzuki, Kayo" uniqKey="Suzuki K" first="Kayo" last="Suzuki">Kayo Suzuki</name><affiliation><nlm:aff id="af1-mco-01-05-0903">Department of Orthopaedic Surgery, Faculty of Medicine, University of Toyama, Toyama, Toyama 930-0194, Japan</nlm:aff></affiliation></author><author><name sortKey="Yasuda, Taketoshi" sort="Yasuda, Taketoshi" uniqKey="Yasuda T" first="Taketoshi" last="Yasuda">Taketoshi Yasuda</name><affiliation><nlm:aff id="af1-mco-01-05-0903">Department of Orthopaedic Surgery, Faculty of Medicine, University of Toyama, Toyama, Toyama 930-0194, Japan</nlm:aff></affiliation></author><author><name sortKey="Hori, Takeshi" sort="Hori, Takeshi" uniqKey="Hori T" first="Takeshi" last="Hori">Takeshi Hori</name><affiliation><nlm:aff id="af1-mco-01-05-0903">Department of Orthopaedic Surgery, Faculty of Medicine, University of Toyama, Toyama, Toyama 930-0194, Japan</nlm:aff></affiliation></author><author><name sortKey="Hachinoda, Jun" sort="Hachinoda, Jun" uniqKey="Hachinoda J" first="Jun" last="Hachinoda">Jun Hachinoda</name><affiliation><nlm:aff id="af2-mco-01-05-0903">Department of Orthopaedic Surgery, Takaoka City Hospital, Takaoka, Toyama 933-0816, Japan</nlm:aff></affiliation></author><author><name sortKey="Kanamori, Masahiko" sort="Kanamori, Masahiko" uniqKey="Kanamori M" first="Masahiko" last="Kanamori">Masahiko Kanamori</name><affiliation><nlm:aff id="af3-mco-01-05-0903">Department of Human Science, Faculty of Medicine, University of Toyama, Toyama, Toyama 930-0194, Japan</nlm:aff></affiliation></author><author><name sortKey="Kimura, Tomoatsu" sort="Kimura, Tomoatsu" uniqKey="Kimura T" first="Tomoatsu" last="Kimura">Tomoatsu Kimura</name><affiliation><nlm:aff id="af1-mco-01-05-0903">Department of Orthopaedic Surgery, Faculty of Medicine, University of Toyama, Toyama, Toyama 930-0194, Japan</nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">24649268</idno><idno type="pmc">3915322</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3915322</idno><idno type="RBID">PMC:3915322</idno><idno type="doi">10.3892/mco.2013.141</idno><date when="2013">2013</date><idno type="wicri:Area/Pmc/Corpus">004212</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">004212</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Cutaneous angiosarcoma of the buttock complicated by severe thrombocytopenia: A case report</title><author><name sortKey="Nagao, Kaoru" sort="Nagao, Kaoru" uniqKey="Nagao K" first="Kaoru" last="Nagao">Kaoru Nagao</name><affiliation><nlm:aff id="af1-mco-01-05-0903">Department of Orthopaedic Surgery, Faculty of Medicine, University of Toyama, Toyama, Toyama 930-0194, Japan</nlm:aff></affiliation></author><author><name sortKey="Suzuki, Kayo" sort="Suzuki, Kayo" uniqKey="Suzuki K" first="Kayo" last="Suzuki">Kayo Suzuki</name><affiliation><nlm:aff id="af1-mco-01-05-0903">Department of Orthopaedic Surgery, Faculty of Medicine, University of Toyama, Toyama, Toyama 930-0194, Japan</nlm:aff></affiliation></author><author><name sortKey="Yasuda, Taketoshi" sort="Yasuda, Taketoshi" uniqKey="Yasuda T" first="Taketoshi" last="Yasuda">Taketoshi Yasuda</name><affiliation><nlm:aff id="af1-mco-01-05-0903">Department of Orthopaedic Surgery, Faculty of Medicine, University of Toyama, Toyama, Toyama 930-0194, Japan</nlm:aff></affiliation></author><author><name sortKey="Hori, Takeshi" sort="Hori, Takeshi" uniqKey="Hori T" first="Takeshi" last="Hori">Takeshi Hori</name><affiliation><nlm:aff id="af1-mco-01-05-0903">Department of Orthopaedic Surgery, Faculty of Medicine, University of Toyama, Toyama, Toyama 930-0194, Japan</nlm:aff></affiliation></author><author><name sortKey="Hachinoda, Jun" sort="Hachinoda, Jun" uniqKey="Hachinoda J" first="Jun" last="Hachinoda">Jun Hachinoda</name><affiliation><nlm:aff id="af2-mco-01-05-0903">Department of Orthopaedic Surgery, Takaoka City Hospital, Takaoka, Toyama 933-0816, Japan</nlm:aff></affiliation></author><author><name sortKey="Kanamori, Masahiko" sort="Kanamori, Masahiko" uniqKey="Kanamori M" first="Masahiko" last="Kanamori">Masahiko Kanamori</name><affiliation><nlm:aff id="af3-mco-01-05-0903">Department of Human Science, Faculty of Medicine, University of Toyama, Toyama, Toyama 930-0194, Japan</nlm:aff></affiliation></author><author><name sortKey="Kimura, Tomoatsu" sort="Kimura, Tomoatsu" uniqKey="Kimura T" first="Tomoatsu" last="Kimura">Tomoatsu Kimura</name><affiliation><nlm:aff id="af1-mco-01-05-0903">Department of Orthopaedic Surgery, Faculty of Medicine, University of Toyama, Toyama, Toyama 930-0194, Japan</nlm:aff></affiliation></author></analytic><series><title level="j">Molecular and Clinical Oncology</title><idno type="ISSN">2049-9450</idno><idno type="eISSN">2049-9469</idno><imprint><date when="2013">2013</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Angiosarcoma (AS) is an aggressive, malignant endothelial cell tumor of vascular or lymphatic origin, the presentation and clinical behavior of which may vary according to its location. This is the case report of a 56-year-old woman with cutaneous angiosarcoma (CAS) of the buttock complicated by severe thrombocytopenia. A review of the literature revealed that only nine cases of CAS with thrombocytopenia have been previously reported. The prognosis of CAS complicated by thrombocytopenia is poor, even after treatment with combined chemotherapy and radiotherapy (RT). The composite karyotype was 46,XX,t(12;20)(p13;p11.2)[3]/47,X,add(X)(q13),del(6)(q?),add(12)(p13),−21,+2mar[2]/45,XX,der(1)add(1)(p36.3)del(1)(q41),−20[1]/46,XX[13]. Only 13 cytogenetic cases of AS, including the present case, have been reported in the English literature thus far. 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<front><journal-meta><journal-id journal-id-type="nlm-ta">Mol Clin Oncol</journal-id><journal-id journal-id-type="iso-abbrev">Mol Clin Oncol</journal-id><journal-id journal-id-type="publisher-id">MCO</journal-id><journal-title-group><journal-title>Molecular and Clinical Oncology</journal-title></journal-title-group><issn pub-type="ppub">2049-9450</issn><issn pub-type="epub">2049-9469</issn><publisher><publisher-name>D.A. Spandidos</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">24649268</article-id><article-id pub-id-type="pmc">3915322</article-id><article-id pub-id-type="doi">10.3892/mco.2013.141</article-id><article-id pub-id-type="publisher-id">mco-01-05-0903</article-id><article-categories><subj-group subj-group-type="heading"><subject>Articles</subject></subj-group></article-categories><title-group><article-title>Cutaneous angiosarcoma of the buttock complicated by severe thrombocytopenia: A case report</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>NAGAO</surname><given-names>KAORU</given-names></name><xref ref-type="aff" rid="af1-mco-01-05-0903">1</xref></contrib><contrib contrib-type="author"><name><surname>SUZUKI</surname><given-names>KAYO</given-names></name><xref ref-type="aff" rid="af1-mco-01-05-0903">1</xref></contrib><contrib contrib-type="author"><name><surname>YASUDA</surname><given-names>TAKETOSHI</given-names></name><xref ref-type="aff" rid="af1-mco-01-05-0903">1</xref><xref ref-type="corresp" rid="c1-mco-01-05-0903"></xref></contrib><contrib contrib-type="author"><name><surname>HORI</surname><given-names>TAKESHI</given-names></name><xref ref-type="aff" rid="af1-mco-01-05-0903">1</xref></contrib><contrib contrib-type="author"><name><surname>HACHINODA</surname><given-names>JUN</given-names></name><xref ref-type="aff" rid="af2-mco-01-05-0903">2</xref></contrib><contrib contrib-type="author"><name><surname>KANAMORI</surname><given-names>MASAHIKO</given-names></name><xref ref-type="aff" rid="af3-mco-01-05-0903">3</xref></contrib><contrib contrib-type="author"><name><surname>KIMURA</surname><given-names>TOMOATSU</given-names></name><xref ref-type="aff" rid="af1-mco-01-05-0903">1</xref></contrib></contrib-group><aff id="af1-mco-01-05-0903"><label>1</label>Department of Orthopaedic Surgery, Faculty of Medicine, University of Toyama, Toyama, Toyama 930-0194, Japan</aff><aff id="af2-mco-01-05-0903"><label>2</label>Department of Orthopaedic Surgery, Takaoka City Hospital, Takaoka, Toyama 933-0816, Japan</aff><aff id="af3-mco-01-05-0903"><label>3</label>Department of Human Science, Faculty of Medicine, University of Toyama, Toyama, Toyama 930-0194, Japan</aff><author-notes><corresp id="c1-mco-01-05-0903">Correspondence to: Dr Taketoshi Yasuda, Department of Orthopaedic Surgery, Faculty of Medicine, University of Toyama, 2630 Sugitani, Toyama, Toyama 930-0194, Japan, E-mail: <email>yasuda@med.u-toyama.ac.jp</email></corresp></author-notes><pub-date pub-type="ppub"><month>9</month><year>2013</year></pub-date><pub-date pub-type="epub"><day>02</day><month>7</month><year>2013</year></pub-date><pub-date pub-type="pmc-release"><day>02</day><month>7</month><year>2013</year></pub-date><pmc-comment> PMC Release delay is 0 months and 0 days and was based on the
<volume>1</volume><issue>5</issue><fpage>903</fpage><lpage>907</lpage><history><date date-type="received"><day>19</day><month>2</month><year>2013</year></date><date date-type="accepted"><day>25</day><month>6</month><year>2013</year></date></history><permissions><copyright-statement>Copyright © 2013, Spandidos Publications</copyright-statement><copyright-year>2013</copyright-year><license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by/3.0"><license-p>This is an open-access article licensed under a Creative Commons Attribution-NonCommercial 3.0 Unported License. The article may be redistributed, reproduced, and reused for non-commercial purposes, provided the original source is properly cited.</license-p></license></permissions><abstract><p>Angiosarcoma (AS) is an aggressive, malignant endothelial cell tumor of vascular or lymphatic origin, the presentation and clinical behavior of which may vary according to its location. This is the case report of a 56-year-old woman with cutaneous angiosarcoma (CAS) of the buttock complicated by severe thrombocytopenia. A review of the literature revealed that only nine cases of CAS with thrombocytopenia have been previously reported. The prognosis of CAS complicated by thrombocytopenia is poor, even after treatment with combined chemotherapy and radiotherapy (RT). The composite karyotype was 46,XX,t(12;20)(p13;p11.2)[3]/47,X,add(X)(q13),del(6)(q?),add(12)(p13),−21,+2mar[2]/45,XX,der(1)add(1)(p36.3)del(1)(q41),−20[1]/46,XX[13]. Only 13 cytogenetic cases of AS, including the present case, have been reported in the English literature thus far. In this case report, the clinical presentation and cytogenetic findings are described and the relevant literature on AS is reviewed.</p></abstract><kwd-group><kwd>cutaneous angiosarcoma</kwd><kwd>thrombocytopenia</kwd><kwd>karyotype</kwd></kwd-group></article-meta></front><body><sec><title>Introduction</title><p>Angiosarcoma (AS) is an aggressive, malignant endothelial cell tumor of vascular or lymphatic origin (<xref rid="b1-mco-01-05-0903" ref-type="bibr">1</xref>). The presentation and clinical behavior of AS may vary, depending on its location. Therefore, ASs are considered as several closely related tumors rather than a single entity and may be divided into several groups. Cutaneous AS (CAS) is a rare aggressive tumor of the scalp and face of elderly patients, which spreads widely, recurring locally with early metastasis (<xref rid="b2-mco-01-05-0903" ref-type="bibr">2</xref>). Cases of thrombocytopenia during tumor progression and metastasis related to intratumor sequestration have been rarely reported (<xref rid="b3-mco-01-05-0903" ref-type="bibr">3</xref>–<xref rid="b8-mco-01-05-0903" ref-type="bibr">8</xref>). The present study describes a case of CAS of the buttock complicated by severe thrombocytopenia and reviews the literature regarding the clinical behavior of this entity. The study was conducted following a clinical research review by our Ethics Committee. The patient was informed that data from the case would be submitted for publication and provided the required consent.</p></sec><sec><title>Case report</title><p>A 56-year-old female sought medical assistance due to a 2-month history of dark reddish eruptions and pain in her left buttock. There was no reported history of disease or trauma. Three discrete eruptions without edema were identified on the skin. The surface was smooth with minor inflammation and the borders were poorly defined. The lesions were tender to palpation. The radiological and laboratory findings were normal. A biopsy was performed and sheets of highly atypical, mitotically active, epithelioid endothelial cells were observed under a light microscope, with formation of irregular sinusoidal vascular channels (<xref rid="f1-mco-01-05-0903" ref-type="fig">Fig. 1</xref>). The lesion was diagnosed as epithelioid hemangioendothelioma based on the histopathological findings.</p><p>Wide surgical resection was performed; however, the patient experienced seven local recurrences over a period of seven years. Whenever the tumor recurred, it was surgically resected. After the third resection the tumor was histopathologically re-evaluated and the diagnosis was changed to AS (<xref rid="f2-mco-01-05-0903" ref-type="fig">Fig. 2</xref>). Immunohistochemically, tumor cells were positive for CD31 and negative for CD34. The specimen was submitted for cytogenetic analysis. Six years after the onset, the tumor had significantly enlarged. Following the seventh surgery, radiotherapy (RT, 60 Gy) and intensive multidrug chemotherapy [MAID (doxorubicin 30 mg/m<sup>2</sup>, ifosfamide 1.5 g/m<sup>2</sup> and dacarbazine 300 mg/m<sup>2</sup>) × 3 days] were administered (<xref rid="b9-mco-01-05-0903" ref-type="bibr">9</xref>). However, one year after the RT the patient developed local recurrence, inguinal lymph node metastasis and multiple lung metastases. Lymphedema also developed due to femoral lymphatic vessel obstruction. Nine years after the initial operation, lymph node dissection and free-skin grafting were performed during the ninth resection procedure. However, the tumor subsequently recurred around the grafted skin and grew progressively, with secondary necrosis and hemorrhage. The hemorrhage persisted for 2 months and the patient developed severe anemia (5.2 g/dl) and thrombocytopenia (52,000/μl). Despite supportive care with hemorrhage control and transfusion of platelets and erythrocytes, anemia and thrombocytopenia continued to progress rapidly. The patient eventually developed disseminated intravascular coagulation and succumbed to the disease 6 weeks after the ninth surgery (9 years and 6 months after the initial consultation).</p><p>Cytogenetic analysis was performed on G-band by trypsin banding using surgical specimens. Standard culture and harvesting procedures were used, as previously described (<xref rid="b10-mco-01-05-0903" ref-type="bibr">10</xref>). The karyotypes were expressed in accordance with the International System for Human Cytogenetic Nomenclature (<xref rid="b11-mco-01-05-0903" ref-type="bibr">11</xref>). Twenty cells were analyzed and the composite karyotype was 46,XX,t(12;20)(p13;p11.2)[3]/47,X,add(X)(q13),del(6)(q?),add(12)(p13),−21,+2mar[2]/45,XX,der(1)add(1)(p36.3)del(1)(q41),−20[1]/46,XX[13] (<xref rid="f3-mco-01-05-0903" ref-type="fig">Fig. 3</xref>).</p></sec><sec sec-type="discussion"><title>Discussion</title><p>ASs are, collectively, one of the rarest types of soft tissue tumors. They comprise <1% of all sarcomas. Among all cases of AS, one third occur in the skin, one fourth in soft tissue and the remainder in other sites. Since the clinical behavior of AS varies depending on its location, ASs are divided into several clinical groups. CAS without lymphedema is the most frequently encountered type of AS. It usually develops after the age of 60 years and more commonly affects females. Due to the fact that AS readily invades the surrounding tissues, complete resection is difficult. The prognosis of CAS is poor, with a 5-year survival rate of 10–34% (<xref rid="b12-mco-01-05-0903" ref-type="bibr">12</xref>–<xref rid="b14-mco-01-05-0903" ref-type="bibr">14</xref>).</p><p>Only 9 cases of CAS with thrombocytopenia, including the present case, have been described in the English literature thus far (<xref rid="b3-mco-01-05-0903" ref-type="bibr">3</xref>–<xref rid="b8-mco-01-05-0903" ref-type="bibr">8</xref>) (<xref rid="tI-mco-01-05-0903" ref-type="table">Table I</xref>). The mean age at presentation was 72.7 years (range, 56–87 years) and it occurred more commonly among males. The tumors were located in the scalp (5/9, 55.6%), face (2/9, 22.2%) and trunk (2/9, 22.2%). The prognosis of CAS complicated by thrombocytopenia is poor, even following treatment with combined chemotherapy and RT. The average period from the initial diagnosis to the development of thrombocytopenia was 4.1 months (range, 0–10 months) and the average survival was 9.7 months (range, 3–36 months). Of the 9 reported cases of CAS with thrombocytopenia (including the present case), all the patients succumbed to the disease within 4 months after the onset of thrombocytopenia. Our patient survived for 76 months subsequent to the initial consultation but succumbed to the disease 2 months following tumor necrosis. These findings suggest that local control of the tumor is critical.</p><p>The present case describes the development of thrombocytopenia in a patient with metastatic AS of the buttock. The mechanism of thrombocytopenia is considered to be related to the Kasabach-Merritt syndrome (KMS) (<xref rid="b15-mco-01-05-0903" ref-type="bibr">15</xref>). KMS has been used to describe various tumors that broadly fit the initial description (<xref rid="b16-mco-01-05-0903" ref-type="bibr">16</xref>), such as thrombocytopenia associated with giant hemangioendothelioma (<xref rid="b5-mco-01-05-0903" ref-type="bibr">5</xref>), intestinal angioma (<xref rid="b17-mco-01-05-0903" ref-type="bibr">17</xref>), hepatic epithelioid hemangioendothelioma (<xref rid="b18-mco-01-05-0903" ref-type="bibr">18</xref>), occult visceral hemangiomatosis (<xref rid="b19-mco-01-05-0903" ref-type="bibr">19</xref>) and AS (<xref rid="b20-mco-01-05-0903" ref-type="bibr">20</xref>). KMS is defined as consumption of platelets within tumors due to the accumulation of blood in the tumor vasculature and platelet accretion to the abnormal vascular endothelium (<xref rid="b16-mco-01-05-0903" ref-type="bibr">16</xref>,<xref rid="b20-mco-01-05-0903" ref-type="bibr">20</xref>). In our case, tumor growth and recurrence led to KMS secondary to hyperplasia of the abnormal blood vessels and continuous bleeding due to tumor necrosis.</p><p>Only a few cases of AS have been cytogenetically investigated (<xref rid="b21-mco-01-05-0903" ref-type="bibr">21</xref>–<xref rid="b29-mco-01-05-0903" ref-type="bibr">29</xref>). The cytogenetic findings of AS are occasionally complex and limited. However, certain cytogenetic changes reported in tumors from different locations revealed similarities (<xref rid="tII-mco-01-05-0903" ref-type="table">Table II</xref>). The most common changes were gain of 5pter-p11, 8p12-qter and 20pter-q12, losses of 4p, 7p15-pter and Y and abnormalities in 22q (<xref rid="b1-mco-01-05-0903" ref-type="bibr">1</xref>). In our case, t(12;20)(p13;p11.2) including a site of 20pter-q12 was observed.</p><p>Treatment of CAS is challenging and an optimal treatment protocol has not yet been determined. The treatment usually consists of surgical excision with wide safety margins and postoperative radiation (<xref rid="b2-mco-01-05-0903" ref-type="bibr">2</xref>,<xref rid="b30-mco-01-05-0903" ref-type="bibr">30</xref>), although radiation monotherapy appears to be insufficient. In addition, the role of chemotherapy remains undetermined and definitive application of adjuvant modalities has not yet been established (<xref rid="b2-mco-01-05-0903" ref-type="bibr">2</xref>,<xref rid="b30-mco-01-05-0903" ref-type="bibr">30</xref>). Additional comprehensive studies on the treatment of CAS are required.</p></sec></body><back><ack><title>Acknowledgements</title><p>This study was supported in part by Grants-in-Aid for Scientific Research (C) 24592227 (KAKENHI).</p></ack><ref-list><title>References</title><ref id="b1-mco-01-05-0903"><label>1</label><element-citation publication-type="book"><person-group person-group-type="author"><name><surname>Weiss</surname><given-names>SW</given-names></name><name><surname>Goldblum</surname><given-names>JR</given-names></name></person-group><article-title>Angiosarcoma</article-title><source>Enzinger and Weiss's Soft Tissue Tumors</source><person-group person-group-type="editor"><name><surname>Weiss</surname><given-names>SW</given-names></name><name><surname>Goldblum</surname><given-names>JR</given-names></name></person-group><publisher-name>Mosby</publisher-name><publisher-loc>St. 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Sheets of highly atypical, mitotically active, epithelioid endothelial cells and irregular sinusoidal vascular channels are present. The initial clinicohistopathological diagnosis was epithelioid hemangioendothelima (hematoxylin and eosin stain; scale bar, 100 μm).</p></caption><graphic xlink:href="MCO-01-05-0903-g00"></graphic></fig><fig id="f2-mco-01-05-0903" orientation="portrait" position="float"><label>Figure 2</label><caption><p>Histological appearance of the third surgical specimen. Sheets of highly pleomorphic endothelial cells and irregular vascular channels filled with erythrocytes are observed. These findings led to the diagnosis of angiosarcoma (hematoxylin and eosin stain; scale bar, 100 μm).</p></caption><graphic xlink:href="MCO-01-05-0903-g01"></graphic></fig><fig id="f3-mco-01-05-0903" orientation="portrait" position="float"><label>Figure 3</label><caption><p>Representative G-banded karyotype. Translocation t(12;20)(p13;p11.2) is identified (arrows).</p></caption><graphic xlink:href="MCO-01-05-0903-g02"></graphic></fig><table-wrap id="tI-mco-01-05-0903" orientation="portrait" position="float"><label>Table I</label><caption><p>Clinical characteristics of cutaneous angiosarcoma with thrombocytopenia.</p></caption><table frame="hsides" rules="groups"><thead><tr><th align="left" valign="bottom" rowspan="1" colspan="1">Case</th><th align="center" valign="bottom" rowspan="1" colspan="1">Age (years)/gender</th><th align="center" valign="bottom" rowspan="1" colspan="1">Location</th><th align="center" valign="bottom" rowspan="1" colspan="1">Treatment</th><th align="center" valign="bottom" rowspan="1" colspan="1">Duration<xref rid="tfn1-mco-01-05-0903" ref-type="table-fn">a</xref> (months)</th><th align="center" valign="bottom" rowspan="1" colspan="1">Outcome (months)</th><th align="center" valign="bottom" rowspan="1" colspan="1">Authors (Refs.)</th></tr></thead><tbody><tr><td align="left" valign="top" rowspan="1" colspan="1">1</td><td align="left" valign="top" rowspan="1" colspan="1">66/F</td><td align="center" valign="top" rowspan="1" colspan="1">Face</td><td align="center" valign="top" rowspan="1" colspan="1">RE</td><td align="right" valign="top" rowspan="1" colspan="1">0</td><td align="left" valign="top" rowspan="1" colspan="1">DOD (3)</td><td align="left" valign="top" rowspan="1" colspan="1">Arcomano <italic>et al</italic>(<xref rid="b3-mco-01-05-0903" ref-type="bibr">3</xref>)</td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">2</td><td align="left" valign="top" rowspan="1" colspan="1">59/M</td><td align="center" valign="top" rowspan="1" colspan="1">Scalp</td><td align="center" valign="top" rowspan="1" colspan="1">CT, RT</td><td align="right" valign="top" rowspan="1" colspan="1">7</td><td align="left" valign="top" rowspan="1" colspan="1">DOD (10)</td><td align="left" valign="top" rowspan="1" colspan="1">Satoh <italic>et al</italic>(<xref rid="b4-mco-01-05-0903" ref-type="bibr">4</xref>)</td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">3</td><td align="left" valign="top" rowspan="1" colspan="1">79/M</td><td align="center" valign="top" rowspan="1" colspan="1">Face</td><td align="center" valign="top" rowspan="1" colspan="1">RT, CT</td><td align="right" valign="top" rowspan="1" colspan="1">3</td><td align="left" valign="top" rowspan="1" colspan="1">DOD (4)</td><td align="left" valign="top" rowspan="1" colspan="1"></td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">4</td><td align="left" valign="top" rowspan="1" colspan="1">79/M</td><td align="center" valign="top" rowspan="1" colspan="1">Chest</td><td align="center" valign="top" rowspan="1" colspan="1">CT</td><td align="right" valign="top" rowspan="1" colspan="1">2</td><td align="left" valign="top" rowspan="1" colspan="1">DOD (6)</td><td align="left" valign="top" rowspan="1" colspan="1"></td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">5</td><td align="left" valign="top" rowspan="1" colspan="1">69/M</td><td align="center" valign="top" rowspan="1" colspan="1">Scalp</td><td align="center" valign="top" rowspan="1" colspan="1">RE, RT, CT</td><td align="right" valign="top" rowspan="1" colspan="1">3</td><td align="left" valign="top" rowspan="1" colspan="1">DOD (4)</td><td align="left" valign="top" rowspan="1" colspan="1">Salameh <italic>et al</italic>(<xref rid="b5-mco-01-05-0903" ref-type="bibr">5</xref>)</td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">6</td><td align="left" valign="top" rowspan="1" colspan="1">67/M</td><td align="center" valign="top" rowspan="1" colspan="1">Scalp</td><td align="center" valign="top" rowspan="1" colspan="1">CT, RT</td><td align="right" valign="top" rowspan="1" colspan="1">10</td><td align="left" valign="top" rowspan="1" colspan="1">DOD (36)</td><td align="left" valign="top" rowspan="1" colspan="1">Imafuku <italic>et al</italic>(<xref rid="b6-mco-01-05-0903" ref-type="bibr">6</xref>)</td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">7</td><td align="left" valign="top" rowspan="1" colspan="1">76/M</td><td align="center" valign="top" rowspan="1" colspan="1">Scalp</td><td align="center" valign="top" rowspan="1" colspan="1">RT, CT</td><td align="right" valign="top" rowspan="1" colspan="1">7</td><td align="left" valign="top" rowspan="1" colspan="1">DOD (9)</td><td align="left" valign="top" rowspan="1" colspan="1">Kluger <italic>et al</italic>(<xref rid="b7-mco-01-05-0903" ref-type="bibr">7</xref>)</td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">8</td><td align="left" valign="top" rowspan="1" colspan="1">87/M</td><td align="center" valign="top" rowspan="1" colspan="1">Scalp</td><td align="center" valign="top" rowspan="1" colspan="1">RT, CT</td><td align="right" valign="top" rowspan="1" colspan="1">1</td><td align="left" valign="top" rowspan="1" colspan="1">DOD (3)</td><td align="left" valign="top" rowspan="1" colspan="1">Tan <italic>et al</italic>(<xref rid="b8-mco-01-05-0903" ref-type="bibr">8</xref>)</td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">9</td><td align="left" valign="top" rowspan="1" colspan="1">56/F</td><td align="center" valign="top" rowspan="1" colspan="1">Buttock</td><td align="center" valign="top" rowspan="1" colspan="1">RE, CT, RT</td><td align="right" valign="top" rowspan="1" colspan="1">72</td><td align="left" valign="top" rowspan="1" colspan="1">DOD (76)</td><td align="left" valign="top" rowspan="1" colspan="1">Present case</td></tr></tbody></table><table-wrap-foot><fn id="tfn1-mco-01-05-0903"><label>a</label><p>Duration indicates the time from the first consultation to the development of thrombocytopenia.</p></fn><fn id="tfn2-mco-01-05-0903"><p>F, female; M, male; RE, resection; CT, chemotherapy; RT, radiotherapy; DOD, dead of disease.</p></fn></table-wrap-foot></table-wrap><table-wrap id="tII-mco-01-05-0903" orientation="portrait" position="float"><label>Table II</label><caption><p>Chromosomal changes in angiosarcoma.</p></caption><table frame="hsides" rules="groups"><thead><tr><th align="left" valign="bottom" rowspan="1" colspan="1">Case</th><th align="center" valign="bottom" rowspan="1" colspan="1">Age (years)/gender</th><th align="center" valign="bottom" rowspan="1" colspan="1">Location</th><th align="center" valign="bottom" rowspan="1" colspan="1">Karyotype (no. of cells)</th><th align="center" valign="bottom" rowspan="1" colspan="1">Authors (Refs.)</th></tr></thead><tbody><tr><td align="left" valign="top" rowspan="1" colspan="1">1</td><td align="center" valign="top" rowspan="1" colspan="1">52/F</td><td align="center" valign="top" rowspan="1" colspan="1">Soft tissue</td><td align="center" valign="top" rowspan="1" colspan="1">41,XY,−3,−6,−6,+der(7)t(6;7)(p21.1;p22),−17,−17,−20[1]/45,XX,−1,+2,−22[1]/45,XX,−6[1]/45,XX,−10[1]/45, XX,−13[1]/45,XX,−14[1]/45,XX,−16[1]/45,XX,−20[1]/45,XX,−22[1]/45,X,−X[1]/46,X,+del(1)(q21), t(6:8) (q22–23;q24),−X[1]/46,XXt(1;12)(p34.1–34.3;q13)[1]/46,XX,t(5;?)(p13;?)[1]/46,X,t(5;19)(q21–22;p13.3), −X,+mar[1]/46,XX,t(10;17)(q11.2;p11.2)[1]/47,XX,t(3;21)(p21;p13),del(7)(q21),+19[1]/47,XX, +3,t(X;10)(q28;q11.2)[1]/48,XX,+2,+7[1]/46,XX[13]</td><td align="center" valign="top" rowspan="1" colspan="1">Kindblom <italic>et al</italic>(<xref rid="b21-mco-01-05-0903" ref-type="bibr">21</xref>)</td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">2</td><td align="center" valign="top" rowspan="1" colspan="1">34/F</td><td align="center" valign="top" rowspan="1" colspan="1">Ovary</td><td align="center" valign="top" rowspan="1" colspan="1">48,XX,t(1;3)(q11;p11),+3,+12</td><td align="center" valign="top" rowspan="1" colspan="1">Fletcher <italic>et al</italic>(<xref rid="b22-mco-01-05-0903" ref-type="bibr">22</xref>)</td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">3</td><td align="center" valign="top" rowspan="1" colspan="1">61/F</td><td align="center" valign="top" rowspan="1" colspan="1">Breast</td><td align="center" valign="top" rowspan="1" colspan="1">47,X,der(X)t(X;9)(q11;q12),t(1;3)(p13;q29),t(1;17)(p13;p13),−2,−2,del(4)(q25q31), +der(4)del(4)(p14)t(2;4)(p13;q35),del(7)(p11),add(8)(q13),+12,der(15)t(5;15)(p11;p11), +der(20)t(6;20)(q13;q23),der(22)t(6;22)(q15;q13)</td><td align="center" valign="top" rowspan="1" colspan="1">Gil-Benso <italic>et al</italic>(<xref rid="b23-mco-01-05-0903" ref-type="bibr">23</xref>)</td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">4</td><td align="center" valign="top" rowspan="1" colspan="1">NA</td><td align="center" valign="top" rowspan="1" colspan="1">Soft tissue</td><td align="center" valign="top" rowspan="1" colspan="1">46,XX,add(1)(p36),t(1;3)(q32;q21),del(2)(p21),del(7)(p15),add(12)(q13),add(17)(q24)</td><td align="center" valign="top" rowspan="1" colspan="1">Van den Berg <italic>et al</italic> 24)</td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">5</td><td align="center" valign="top" rowspan="1" colspan="1">67/M</td><td align="center" valign="top" rowspan="1" colspan="1">Kidney</td><td align="center" valign="top" rowspan="1" colspan="1">46,XY,inv(7)(p15q11)[2]/46–47,X,−Y,del(1)(q25),add(4)(q25),add(5)(q31),+mar1[cp15]/46,XY[3]</td><td align="center" valign="top" rowspan="1" colspan="1">Cerilli <italic>et al</italic>(<xref rid="b25-mco-01-05-0903" ref-type="bibr">25</xref>)</td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">6</td><td align="center" valign="top" rowspan="1" colspan="1">41/M</td><td align="center" valign="top" rowspan="1" colspan="1">Soft tissue</td><td align="center" valign="top" rowspan="1" colspan="1">39–43,XY,der(1;11)(q10;q10),−4,t(5;15)(q11;q24),+der(8)t(8;?17)(p12;q11)add(8)(q24), −9,−11,−13,−17,−17,−18,add(18)(p11),+del(20)(q12–13),+der(?)t(?;10)(?;q22)[cp9]/46,XY[2]</td><td align="center" valign="top" rowspan="1" colspan="1">Schuborg <italic>et al</italic>(<xref rid="b26-mco-01-05-0903" ref-type="bibr">26</xref>)</td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">7</td><td align="center" valign="top" rowspan="1" colspan="1">69/F</td><td align="center" valign="top" rowspan="1" colspan="1">Soft tissue</td><td align="center" valign="top" rowspan="1" colspan="1">45–47,XX,add(8)(q22),+add(22)(q12),inc[cp9]</td><td align="center" valign="top" rowspan="1" colspan="1"></td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">8</td><td align="center" valign="top" rowspan="1" colspan="1">75/M</td><td align="center" valign="top" rowspan="1" colspan="1">Soft tissue</td><td align="center" valign="top" rowspan="1" colspan="1">46,XY,der(7)del(7)(p15)del(7)(q11q22)[3]/47,XY,der(7),+mar[2]/45,X, −Y[13]/47,XY,+8[7]/48,XY,+X,+8[6]</td><td align="center" valign="top" rowspan="1" colspan="1"></td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">9a</td><td align="center" valign="top" rowspan="1" colspan="1">71/F</td><td align="center" valign="top" rowspan="1" colspan="1">Soft tissue</td><td align="center" valign="top" rowspan="1" colspan="1">73–79,XX,−X,add(1)(q11),−3,+5,t(5;14)(q13;p13),+6,+7,+8,+9,+9, (primary) der(9;10)(q10;q10)x2,+11,−13,+20,−21,−22,inc[cp3]/46,XX[11]</td><td align="center" valign="top" rowspan="1" colspan="1"></td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">9b</td><td align="center" valign="top" rowspan="1" colspan="1">71/F</td><td align="center" valign="top" rowspan="1" colspan="1">Soft tissue (recurrence)</td><td align="center" valign="top" rowspan="1" colspan="1">77–79,XXX,add(1),−3,i(5)(q10),t(5;14),+6,+6,+7,der(9;10)x2, +11,add(15)(p11),+16,+20,inc[2]/46,XX[p]</td><td align="center" valign="top" rowspan="1" colspan="1">Wong <italic>et al</italic>(<xref rid="b27-mco-01-05-0903" ref-type="bibr">27</xref>)</td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">10</td><td align="center" valign="top" rowspan="1" colspan="1">34/F</td><td align="center" valign="top" rowspan="1" colspan="1">Nasopharynx</td><td align="center" valign="top" rowspan="1" colspan="1">43,XX,?dup(3)(q21q29),del(4)(p11p16),−8,der(9;17)(q10;q10),−13,add(14)(q32), −16,add(16)(q22),der(18)t(1;18)(q21;q21),−19,−20,add(22)(q13),+r,+mar1,+mar2[4]/43, idem, −14,+20[3]/33–43,idem,−3,−4,−5,−6,−7,−10,−11,−16,−18,−20[cp10]/61–84,XXX,?dup(3),+?dup(3), del(4),+del(4),+5,+7,−8,der(9;17),?i(9)(q10),+10,+11,+12,+14,+15,−16,add(16)x2,−17,add(18), add(19)(p13),−20,add(22),+add(22),+rx2,+mar1×2,+mar2×2[cp6]/46,XX[3]</td><td align="center" valign="top" rowspan="1" colspan="1"></td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">11</td><td align="center" valign="top" rowspan="1" colspan="1">29/M</td><td align="center" valign="top" rowspan="1" colspan="1">Heart</td><td align="center" valign="top" rowspan="1" colspan="1">55,XY,+der(1;17)(q10;q10),+2,+7,+8,+8,+19,+20,+21,+22</td><td align="center" valign="top" rowspan="1" colspan="1">Zu <italic>et al</italic>(<xref rid="b28-mco-01-05-0903" ref-type="bibr">28</xref>)</td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">12</td><td align="center" valign="top" rowspan="1" colspan="1">79/F</td><td align="center" valign="top" rowspan="1" colspan="1">Bone</td><td align="center" valign="top" rowspan="1" colspan="1">46,XX,t(1;14)(p21;q24)[16]/46,XX[3]</td><td align="center" valign="top" rowspan="1" colspan="1">Dunlap <italic>et al</italic>(<xref rid="b29-mco-01-05-0903" ref-type="bibr">29</xref>)</td></tr><tr><td align="left" valign="top" rowspan="1" colspan="1">13</td><td align="center" valign="top" rowspan="1" colspan="1">56/F</td><td align="center" valign="top" rowspan="1" colspan="1">Cutaneous</td><td align="center" valign="top" rowspan="1" colspan="1">46,XX,t(12;20)(p13;p11.2)[3]/47,X,add(X)(q13),del(6)(q?),add(12)(p13),−21, +2mar[2]/45,XX,der(1)add(1)(p36.3)del(1)(q41),−20[2]/46,XX[13]</td><td align="center" valign="top" rowspan="1" colspan="1">Present case</td></tr></tbody></table><table-wrap-foot><fn id="tfn3-mco-01-05-0903"><p>F, female; M, male, NA, not available.</p></fn></table-wrap-foot></table-wrap></floats-group></pmc></record>Pour manipuler ce document sous Unix (Dilib)
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