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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Unresectable Retiform Hemangioendothelioma Treated with External Beam Radiation Therapy and Chemotherapy: A Case Report and Review of the Literature</title><author><name sortKey="Hirsh, Alina Z" sort="Hirsh, Alina Z" uniqKey="Hirsh A" first="Alina Z." last="Hirsh">Alina Z. Hirsh</name><affiliation><nlm:aff id="I1">Stich Radiation Center, Weill Cornell Medical Center, New York, NY 10065, USA</nlm:aff></affiliation></author><author><name sortKey="Yan, Weisi" sort="Yan, Weisi" uniqKey="Yan W" first="Weisi" last="Yan">Weisi Yan</name><affiliation><nlm:aff id="I1">Stich Radiation Center, Weill Cornell Medical Center, New York, NY 10065, USA</nlm:aff></affiliation></author><author><name sortKey="Wei, Lihong" sort="Wei, Lihong" uniqKey="Wei L" first="Lihong" last="Wei">Lihong Wei</name><affiliation><nlm:aff id="I2">Department of Hematology/Oncology, New York Hospital Queens, NY 11355, USA</nlm:aff></affiliation></author><author><name sortKey="Wernicke, A Gabriella" sort="Wernicke, A Gabriella" uniqKey="Wernicke A" first="A. Gabriella" last="Wernicke">A. Gabriella Wernicke</name><affiliation><nlm:aff id="I1">Stich Radiation Center, Weill Cornell Medical Center, New York, NY 10065, USA</nlm:aff></affiliation></author><author><name sortKey="Parashar, Bhupesh" sort="Parashar, Bhupesh" uniqKey="Parashar B" first="Bhupesh" last="Parashar">Bhupesh Parashar</name><affiliation><nlm:aff id="I1">Stich Radiation Center, Weill Cornell Medical Center, New York, NY 10065, USA</nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">20936126</idno><idno type="pmc">2948930</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC2948930</idno><idno type="RBID">PMC:2948930</idno><idno type="doi">10.1155/2010/756246</idno><date when="2010">2010</date><idno type="wicri:Area/Pmc/Corpus">002401</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">002401</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Unresectable Retiform Hemangioendothelioma Treated with External Beam Radiation Therapy and Chemotherapy: A Case Report and Review of the Literature</title><author><name sortKey="Hirsh, Alina Z" sort="Hirsh, Alina Z" uniqKey="Hirsh A" first="Alina Z." last="Hirsh">Alina Z. Hirsh</name><affiliation><nlm:aff id="I1">Stich Radiation Center, Weill Cornell Medical Center, New York, NY 10065, USA</nlm:aff></affiliation></author><author><name sortKey="Yan, Weisi" sort="Yan, Weisi" uniqKey="Yan W" first="Weisi" last="Yan">Weisi Yan</name><affiliation><nlm:aff id="I1">Stich Radiation Center, Weill Cornell Medical Center, New York, NY 10065, USA</nlm:aff></affiliation></author><author><name sortKey="Wei, Lihong" sort="Wei, Lihong" uniqKey="Wei L" first="Lihong" last="Wei">Lihong Wei</name><affiliation><nlm:aff id="I2">Department of Hematology/Oncology, New York Hospital Queens, NY 11355, USA</nlm:aff></affiliation></author><author><name sortKey="Wernicke, A Gabriella" sort="Wernicke, A Gabriella" uniqKey="Wernicke A" first="A. Gabriella" last="Wernicke">A. Gabriella Wernicke</name><affiliation><nlm:aff id="I1">Stich Radiation Center, Weill Cornell Medical Center, New York, NY 10065, USA</nlm:aff></affiliation></author><author><name sortKey="Parashar, Bhupesh" sort="Parashar, Bhupesh" uniqKey="Parashar B" first="Bhupesh" last="Parashar">Bhupesh Parashar</name><affiliation><nlm:aff id="I1">Stich Radiation Center, Weill Cornell Medical Center, New York, NY 10065, USA</nlm:aff></affiliation></author></analytic><series><title level="j">Sarcoma</title><idno type="ISSN">1357-714X</idno><idno type="eISSN">1369-1643</idno><imprint><date when="2010">2010</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Retiform hemangioendothelioma (RH) is an infrequently encountered vascular neoplasm of intermediate or borderline malignancy. Treatment of RH is controversial. We present a case of a 44-year-old Asian male presenting with an unresectable RH of the pelvis. The patient was treated with concurrent low-dose Cisplatin and External beam Radiation (4140cGy in 180cGy per fraction). This is the first report of a clinical complete response and a long-term local control of this rare tumor. This has significant clinical implication, since it gives the first evidence of treatment of this rare tumor using concurrent low-dose chemotherapy and radiation.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Calonje, E" uniqKey="Calonje E">E Calonje</name></author><author><name sortKey="Fletcher, Cdm" uniqKey="Fletcher C">CDM Fletcher</name></author><author><name sortKey="Wilson Jones, E" uniqKey="Wilson Jones E">E Wilson-Jones</name></author><author><name sortKey="Rosai, J" uniqKey="Rosai J">J Rosai</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Tan, D" uniqKey="Tan D">D Tan</name></author><author><name sortKey="Kraybill, W" uniqKey="Kraybill W">W Kraybill</name></author><author><name sortKey="Cheney, Rt" uniqKey="Cheney R">RT Cheney</name></author><author><name sortKey="Khoury, T" uniqKey="Khoury T">T Khoury</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Duke, D" uniqKey="Duke D">D Duke</name></author><author><name sortKey="Dvorak, Am" uniqKey="Dvorak A">AM Dvorak</name></author><author><name sortKey="Harris, Tj" uniqKey="Harris T">TJ Harris</name></author><author><name sortKey="Cohen, Lm" uniqKey="Cohen L">LM Cohen</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Mentzel, T" uniqKey="Mentzel T">T Mentzel</name></author><author><name sortKey="Stengel, B" uniqKey="Stengel B">B Stengel</name></author><author><name sortKey="Katenkamp, D" uniqKey="Katenkamp D">D Katenkamp</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Aditya, Gs" uniqKey="Aditya G">GS Aditya</name></author><author><name sortKey="Santosh, V" uniqKey="Santosh V">V Santosh</name></author><author><name sortKey="Yasha, Tc" uniqKey="Yasha T">TC Yasha</name></author><author><name sortKey="Shankar, Sk" uniqKey="Shankar S">SK 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Sanz-Trelles</name></author><author><name sortKey="Rodrigo Fernandez, I" uniqKey="Rodrigo Fernandez I">I Rodrigo-Fernandez</name></author><author><name sortKey="Ayala Carbonero, A" uniqKey="Ayala Carbonero A">A Ayala-Carbonero</name></author><author><name sortKey="Contreras Rubio, F" uniqKey="Contreras Rubio F">F Contreras-Rubio</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Darouti, Me" uniqKey="Darouti M">ME Darouti</name></author><author><name sortKey="Marzouk, Sa" uniqKey="Marzouk S">SA Marzouk</name></author><author><name sortKey="Sobhi, Rm" uniqKey="Sobhi R">RM Sobhi</name></author><author><name sortKey="Bassiouni, Da" uniqKey="Bassiouni D">DA Bassiouni</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Fukunaga, M" uniqKey="Fukunaga M">M Fukunaga</name></author><author><name sortKey="Endo, Y" uniqKey="Endo Y">Y Endo</name></author><author><name sortKey="Masui, F" uniqKey="Masui F">F Masui</name></author><author><name sortKey="Yoshikawa, T" uniqKey="Yoshikawa T">T Yoshikawa</name></author><author><name sortKey="Ishikawa, E" uniqKey="Ishikawa E">E Ishikawa</name></author><author><name sortKey="Ushigome, S" uniqKey="Ushigome S">S Ushigome</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Dufau, Jp" uniqKey="Dufau J">JP Dufau</name></author><author><name sortKey="Pierre, C" uniqKey="Pierre C">C Pierre</name></author><author><name sortKey="De Saint Maur, P P" uniqKey="De Saint Maur P">P-P De Saint Maur</name></author><author><name sortKey="Bellavoir, A" uniqKey="Bellavoir A">A Bellavoir</name></author><author><name sortKey="Gros, P" uniqKey="Gros P">P Gros</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Brennan, Mf" uniqKey="Brennan M">MF Brennan</name></author><author><name sortKey="Alektiar, Km" uniqKey="Alektiar K">KM Alektiar</name></author><author><name sortKey="Maki, Rg" uniqKey="Maki R">RG Maki</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Parsons, A" uniqKey="Parsons A">A Parsons</name></author><author><name sortKey="Sheehan, Dj" uniqKey="Sheehan D">DJ Sheehan</name></author><author><name sortKey="Sangueza, Op" uniqKey="Sangueza O">OP Sangueza</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">Sarcoma</journal-id><journal-id journal-id-type="iso-abbrev">Sarcoma</journal-id><journal-id journal-id-type="publisher-id">SRCM</journal-id><journal-title-group><journal-title>Sarcoma</journal-title></journal-title-group><issn pub-type="ppub">1357-714X</issn><issn pub-type="epub">1369-1643</issn><publisher><publisher-name>Hindawi Publishing Corporation</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">20936126</article-id><article-id pub-id-type="pmc">2948930</article-id><article-id pub-id-type="doi">10.1155/2010/756246</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>Unresectable Retiform Hemangioendothelioma Treated with External Beam Radiation Therapy and Chemotherapy: A Case Report and Review of the Literature</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Hirsh</surname><given-names>Alina Z.</given-names></name><xref ref-type="aff" rid="I1"><sup>1</sup></xref></contrib><contrib contrib-type="author"><name><surname>Yan</surname><given-names>Weisi</given-names></name><xref ref-type="aff" rid="I1"><sup>1</sup></xref></contrib><contrib contrib-type="author"><name><surname>Wei</surname><given-names>Lihong</given-names></name><xref ref-type="aff" rid="I2"><sup>2</sup></xref></contrib><contrib contrib-type="author"><name><surname>Wernicke</surname><given-names>A. Gabriella</given-names></name><xref ref-type="aff" rid="I1"><sup>1</sup></xref></contrib><contrib contrib-type="author"><name><surname>Parashar</surname><given-names>Bhupesh</given-names></name><xref ref-type="aff" rid="I1"><sup>1</sup></xref><xref ref-type="corresp" rid="cor1">*</xref></contrib></contrib-group><aff id="I1"><sup>1</sup>Stich Radiation Center, Weill Cornell Medical Center, New York, NY 10065, USA</aff><aff id="I2"><sup>2</sup>Department of Hematology/Oncology, New York Hospital Queens, NY 11355, USA</aff><author-notes><corresp id="cor1">*Bhupesh Parashar: <email>bup9001@med.cornell.edu</email></corresp><fn fn-type="other"><p>Academic Editor: Alberto Pappo</p></fn></author-notes><pub-date pub-type="ppub"><year>2010</year></pub-date><pub-date pub-type="epub"><day>26</day><month>9</month><year>2010</year></pub-date><volume>2010</volume><elocation-id>756246</elocation-id><history><date date-type="received"><day>19</day><month>4</month><year>2010</year></date><date date-type="rev-recd"><day>15</day><month>8</month><year>2010</year></date><date date-type="accepted"><day>16</day><month>8</month><year>2010</year></date></history><permissions><copyright-statement>Copyright © 2010 Alina Z. Hirsh et al.</copyright-statement><copyright-year>2010</copyright-year><license xlink:href="https://creativecommons.org/licenses/by/3.0/"><license-p>This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p></license></permissions><abstract><p>Retiform hemangioendothelioma (RH) is an infrequently encountered vascular neoplasm of intermediate or borderline malignancy. Treatment of RH is controversial. We present a case of a 44-year-old Asian male presenting with an unresectable RH of the pelvis. The patient was treated with concurrent low-dose Cisplatin and External beam Radiation (4140cGy in 180cGy per fraction). This is the first report of a clinical complete response and a long-term local control of this rare tumor. This has significant clinical implication, since it gives the first evidence of treatment of this rare tumor using concurrent low-dose chemotherapy and radiation.</p></abstract></article-meta></front><body><sec id="sec1"><title>1. Introduction</title><p>Retiform hemangioendothelioma (RH) is an infrequently encountered vascular neoplasm of intermediate or borderline malignancy. Treatment of RH is controversial though most reports have described surgical resection followed by adjuvant radiation (RT) with or without chemotherapy. We describe a case report of a 44-year-old Asian male presenting with an unresectable RH of the pelvis and successful treated with concurrent chemotherapy and radiation.</p></sec><sec id="sec2"><title>2. Case Report</title><p>A 44 year old male presented with one year history of slowly enlarging tumor of the right medial thigh and scrotum and extending to the left groin (<xref ref-type="fig" rid="fig1">Figure 1(a)</xref>). The lesion was asymptomatic in the beginning and the skin was intact. Gradually the skin over the right inguinal area eroded and the lesion started causing discomfort which became progressively worse. The patient's previous medical history was unremarkable. Laboratory examinations including HIV status were normal. MRI of the right hip showed abnormal fluid-like density ill-defined in nature in the anterior right groin anterior to the pectineus muscle and dissecting into the medial proximal right thigh. There was no clinical or radiological evidence of regional lymph node involvement. Computed tomography scans of the chest and abdomen showed no evidence of metastatic disease. Histological examination of the biopsy specimen revealed numerous anatomizing vascular channels in the dermis. The vascular channels were composed of endothelial cells, some of which had a hobnail appearance. ICC stain for CD31 was positive and HHV8 was negative. Histological diagnosis of hemangioendothelioma with retiform features was established. The patient was initially evaluated for surgical resection but was deemed unresectable because of extent of the disease. A decision was made to treat the patient with chemoradiation and reevaluation after 4140cGy. A 3-dimensional plan was generated to treat the pelvis, after contouring the Gross tumor (based on pretreatment CT and MR images) plus a 2 cm margin (Figures <xref ref-type="fig" rid="fig1">1(b) </xref>and <xref ref-type="fig" rid="fig1">1(c)</xref>). A total dose of 4140cGy in 180cGy per fraction was delivered with an intention of attempt at surgical resection. Weekly Cisplatin (30 mg/m<sup>2</sup>) was given concomitantly with radiation. The patient tolerated the treatment well and had an excellent clinical response (<xref ref-type="fig" rid="fig1">Figure 1(d)</xref>). Reevaluation at 4140cGy revealed a minimal radiological response and it was decided to abort treatment at the time because of tumor unresectibility. The patient was followed with a repeat biopsy 8 weeks after RT. Biopsy showed an excellent response but some residual disease. However, a 6-month followup revealed a complete clinical response. The patient has been disease-free at 36 months.</p></sec><sec id="sec3"><title>3. Discussion</title><p>Retiform hemangioendothelioma (RH) is an infrequently encountered vascular neoplasm of intermediate or borderline malignancy that has been classified as a distinct type of low-grade cutaneous angiosarcoma. Up to date, a total of 25 cases were described in the literature [<xref rid="B1" ref-type="bibr">1</xref>–<xref rid="B11" ref-type="bibr">11</xref>]. First, fifteen cases were described by Calonje et al. in 1994 [<xref rid="B1" ref-type="bibr">1</xref>]. It has been proposed by Calonje that in the past examples of the tumor he designated as RH have been diagnosed as angiosarcoma. It is important to distinguish RH from cutaneous angiosarcoma (CA) as the clinical course is quite different. CA can arise in the context of lymphedema (Stewart-Treves syndrome) so can RH (see above) and can be radiation-induced. Cutaneous angiosarcoma has high incidence of recurrence and metastasis and overall carries poor prognosis with high mortality. In comparison, RH has a high local recurrence rate, however, rarely metastasizes and there have been no tumor-related deaths reported up to date [<xref rid="B1" ref-type="bibr">1</xref>–<xref rid="B11" ref-type="bibr">11</xref>]. Histology is the only way to reliably differentiate RH from CA. Histologically, in CA, the infiltrative pattern is much more disorganized, with sinusoidal or sieve collagen bundles and vascular spaces are more irregular and jagged. Most importantly, even in well-differentiated CA, mitotic figures, cytologic atypia, variation in cellular size, and multilayering can be found [<xref rid="B1" ref-type="bibr">1</xref>, <xref rid="B12" ref-type="bibr">12</xref>], while there is almost no cytologic atypia and no mitotic activity in RH.</p><p>Morphologically, RH presents as exophytic mass or nodule or a plaque, dermal or subcutaneous, that grows slowly. Most tumors present in young to middle-aged adults (Range 9–78 years) [<xref rid="B2" ref-type="bibr">2</xref>]. Most lesions are single though one case of multiple tumors developing in different anatomic sites (trunk and extremities) has been described [<xref rid="B3" ref-type="bibr">3</xref>]. One case of regional lymph node metastasis [<xref rid="B1" ref-type="bibr">1</xref>] and one soft-tissue metastasis has been reported [<xref rid="B4" ref-type="bibr">4</xref>]. There is female predominance 2 : 1 with size range of 1 to 30 cm. Duration of the disease ranged from 2 months to several years. No distant metastases or tumor-related deaths have been reported; however, RH recurs in almost half of the cases. Often, multiple recurrences are observed. Recurrences are observed ranging from months to several years after original presentation. Etiology is unknown; however, an association between RH and human herpes virus type 8 [<xref rid="B6" ref-type="bibr">6</xref>], lymphedema [<xref rid="B1" ref-type="bibr">1</xref>], previous radiotherapy [<xref rid="B1" ref-type="bibr">1</xref>, <xref rid="B6" ref-type="bibr">6</xref>], and noncutaneous malignant neoplasms [<xref rid="B1" ref-type="bibr">1</xref>, <xref rid="B6" ref-type="bibr">6</xref>, <xref rid="B7" ref-type="bibr">7</xref>, <xref rid="B11" ref-type="bibr">11</xref>] has been reported but not clearly established. </p><p>Clinically, RH lesions could be located in the dermis (predominantly) with extension into the subcutaneous tissues or striated muscle as was also reported as in our case [<xref rid="B1" ref-type="bibr">1</xref>]. A characteristic microscopic feature is the presence of elongated, arborizing, thin-walled blood vessels extending between collagen bundles in a retiform pattern, reminiscent of the architecture of normal rete testis [<xref rid="B1" ref-type="bibr">1</xref>]. In some areas, a prominent lymphocytic infiltrate is present. Some areas of the tumor are solid and are composed of epitheloid or spindle cells and dilated vascular channels sometimes with intraluminal papillary projections. Cytologic atypia is minimal and few or no mitotic figures are seen [<xref rid="B1" ref-type="bibr">1</xref>, <xref rid="B7" ref-type="bibr">7</xref>]. Immunohistochemically, neoplastic cells are positive for CD 31, CD 34, factor VIII-related antigen, and bound <italic>Ulex earopaeus agglutinin </italic>(UEA) (7). The spindle cells are positive for UEA-1 and CD 31 but not for FVIII-Rag and CD34 [<xref rid="B1" ref-type="bibr">1</xref>]. RH may rarely express D2-40, but it does not usually express VEGR-3, markers of endothelium of lymphatic vessels [<xref rid="B13" ref-type="bibr">13</xref>]. In summary, based on the staining patterns, it has been suggested that RH is a vascular entity which usually does not have lymphatic differentiation [<xref rid="B13" ref-type="bibr">13</xref>].</p><p>Other vascular tumors that morphologically can present similarly to RH and need to be differentiated from RH include Dabska's tumor, lymphoma, dermatofibrosarcoma protuberans, hemangioma, bacillary angiomatosis, cutaneous metastases, blue-rubber bleb nevus syndrome, Kaposi's sarcoma, targetoid hemosiderotic hemangioma, hobnail hemangioma, epitheloid hemangioendothelioma, and polymorphous hemangioendothelioma of lymph node [<xref rid="B1" ref-type="bibr">1</xref>, <xref rid="B2" ref-type="bibr">2</xref>, <xref rid="B7" ref-type="bibr">7</xref>, <xref rid="B9" ref-type="bibr">9</xref>]. Even though some histological overlap exists, each of the neoplasms has their unique features [<xref rid="B1" ref-type="bibr">1</xref>, <xref rid="B2" ref-type="bibr">2</xref>, <xref rid="B8" ref-type="bibr">8</xref>].</p><p> Treatment of choice for RH has been surgical excision with histopathologically tumor-free margins. In the cases reported up to date, patients were treated initially by surgical excision. Approximately 50% of the cases recurred [<xref rid="B1" ref-type="bibr">1</xref>–<xref rid="B13" ref-type="bibr">13</xref>]. Amputation of hand, disarticulation of toe, partial penectomy, and amputation of finger has been described [<xref rid="B1" ref-type="bibr">1</xref>]. Radiotherapy has been reported to be successfully administered as adjuvant treatment for local as well as regional nodal RH recurrences [<xref rid="B1" ref-type="bibr">1</xref>, <xref rid="B6" ref-type="bibr">6</xref>]. In addition, adjuvant immunotherapy with recombinant interferon alpha has been tried. As per our knowledge, our report is the first reported successful treatment of RH with low-dose cisplatin and moderate RT without surgical resection. 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The skin healed within 4–6 weeks of completion of RT.</p></caption><graphic xlink:href="SRCM2010-756246.001"></graphic></fig></floats-group></pmc></record>Pour manipuler ce document sous Unix (Dilib)
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