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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Recurrent cutaneous angiosarcoma of the ear masquerading as atypical fibroxanthoma</title><author><name sortKey="Gonzalez, Cristian D" sort="Gonzalez, Cristian D" uniqKey="Gonzalez C" first="Cristian D." last="Gonzalez">Cristian D. Gonzalez</name><affiliation><nlm:aff id="aff1">Department of Dermatology, University of Utah School of Medicine, Salt Lake City, Utah</nlm:aff></affiliation></author><author><name sortKey="Hawkes, Jason E" sort="Hawkes, Jason E" uniqKey="Hawkes J" first="Jason E." last="Hawkes">Jason E. Hawkes</name><affiliation><nlm:aff id="aff1">Department of Dermatology, University of Utah School of Medicine, Salt Lake City, Utah</nlm:aff></affiliation></author><author><name sortKey="Bowles, Tawnya L" sort="Bowles, Tawnya L" uniqKey="Bowles T" first="Tawnya L." last="Bowles">Tawnya L. Bowles</name><affiliation><nlm:aff id="aff2">Melanoma and Cutaneous Oncology Program, Huntsman Cancer Institute, University of Utah, Salt Lake City, Utah</nlm:aff></affiliation><affiliation><nlm:aff id="aff3">Intermountain Health Care, Salt Lake City, Utah</nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">27981214</idno><idno type="pmc">5148779</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC5148779</idno><idno type="RBID">PMC:5148779</idno><idno type="doi">10.1016/j.jdcr.2016.09.005</idno><date when="2016">2016</date><idno type="wicri:Area/Pmc/Corpus">002329</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">002329</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Recurrent cutaneous angiosarcoma of the ear masquerading as atypical fibroxanthoma</title><author><name sortKey="Gonzalez, Cristian D" sort="Gonzalez, Cristian D" uniqKey="Gonzalez C" first="Cristian D." last="Gonzalez">Cristian D. Gonzalez</name><affiliation><nlm:aff id="aff1">Department of Dermatology, University of Utah School of Medicine, Salt Lake City, Utah</nlm:aff></affiliation></author><author><name sortKey="Hawkes, Jason E" sort="Hawkes, Jason E" uniqKey="Hawkes J" first="Jason E." last="Hawkes">Jason E. Hawkes</name><affiliation><nlm:aff id="aff1">Department of Dermatology, University of Utah School of Medicine, Salt Lake City, Utah</nlm:aff></affiliation></author><author><name sortKey="Bowles, Tawnya L" sort="Bowles, Tawnya L" uniqKey="Bowles T" first="Tawnya L." last="Bowles">Tawnya L. Bowles</name><affiliation><nlm:aff id="aff2">Melanoma and Cutaneous Oncology Program, Huntsman Cancer Institute, University of Utah, Salt Lake City, Utah</nlm:aff></affiliation><affiliation><nlm:aff id="aff3">Intermountain Health Care, Salt Lake City, Utah</nlm:aff></affiliation></author></analytic><series><title level="j">JAAD Case Reports</title><idno type="eISSN">2352-5126</idno><imprint><date when="2016">2016</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Albores Saavedra, J" uniqKey="Albores Saavedra J">J. Albores-saavedra</name></author><author><name sortKey="Schwartz, A M" uniqKey="Schwartz A">A.M. Schwartz</name></author><author><name sortKey="Henson, D E" uniqKey="Henson D">D.E. Henson</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Patel, S H" uniqKey="Patel S">S.H. Patel</name></author><author><name sortKey="Hayden, R E" uniqKey="Hayden R">R.E. Hayden</name></author><author><name sortKey="Hinni, M L" uniqKey="Hinni M">M.L. Hinni</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Barhmi, I" uniqKey="Barhmi I">I. Barhmi</name></author><author><name sortKey="Abada, R" uniqKey="Abada R">R. Abada</name></author><author><name sortKey="Roubal, M" uniqKey="Roubal M">M. Roubal</name></author><author><name sortKey="Mahtar, M" uniqKey="Mahtar M">M. Mahtar</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Leighton, S E" uniqKey="Leighton S">S.E. Leighton</name></author><author><name sortKey="Levine, T P" uniqKey="Levine T">T.P. Levine</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Penel, N" uniqKey="Penel N">N. Penel</name></author><author><name sortKey="Italiano, A" uniqKey="Italiano A">A. Italiano</name></author><author><name sortKey="Ray Coquard, I" uniqKey="Ray Coquard I">I. Ray-coquard</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Chow, W" uniqKey="Chow W">W. Chow</name></author><author><name sortKey="Amaya, C N" uniqKey="Amaya C">C.N. Amaya</name></author><author><name sortKey="Rains, S" uniqKey="Rains S">S. Rains</name></author><author><name sortKey="Chow, M" uniqKey="Chow M">M. Chow</name></author><author><name sortKey="Dickerson, E B" uniqKey="Dickerson E">E.B. Dickerson</name></author><author><name sortKey="Bryan, B A" uniqKey="Bryan B">B.A. Bryan</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Wada, M" uniqKey="Wada M">M. Wada</name></author><author><name sortKey="Horinaka, M" uniqKey="Horinaka M">M. Horinaka</name></author><author><name sortKey="Yasuda, S" uniqKey="Yasuda S">S. Yasuda</name></author><author><name sortKey="Masuzawa, M" uniqKey="Masuzawa M">M. Masuzawa</name></author><author><name sortKey="Sakai, T" uniqKey="Sakai T">T. Sakai</name></author><author><name sortKey="Katoh, N" uniqKey="Katoh N">N. Katoh</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Hollmig, S T" uniqKey="Hollmig S">S.T. Hollmig</name></author><author><name sortKey="Sachdev, R" uniqKey="Sachdev R">R. Sachdev</name></author><author><name sortKey="Cockerell, C J" uniqKey="Cockerell C">C.J. Cockerell</name></author><author><name sortKey="Posten, W" uniqKey="Posten W">W. Posten</name></author><author><name sortKey="Chiang, M" uniqKey="Chiang M">M. Chiang</name></author><author><name sortKey="Kim, J" uniqKey="Kim J">J. Kim</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Birdsey, G M" uniqKey="Birdsey G">G.M. Birdsey</name></author><author><name sortKey="Dryden, N H" uniqKey="Dryden N">N.H. Dryden</name></author><author><name sortKey="Shah, A V" uniqKey="Shah A">A.V. Shah</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Sullivan, H C" uniqKey="Sullivan H">H.C. Sullivan</name></author><author><name sortKey="Edgar, M A" uniqKey="Edgar M">M.A. Edgar</name></author><author><name sortKey="Cohen, C" uniqKey="Cohen C">C. Cohen</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">JAAD Case Rep</journal-id><journal-id journal-id-type="iso-abbrev">JAAD Case Rep</journal-id><journal-title-group><journal-title>JAAD Case Reports</journal-title></journal-title-group><issn pub-type="epub">2352-5126</issn><publisher><publisher-name>Elsevier</publisher-name></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">27981214</article-id><article-id pub-id-type="pmc">5148779</article-id><article-id pub-id-type="publisher-id">S2352-5126(16)30105-9</article-id><article-id pub-id-type="doi">10.1016/j.jdcr.2016.09.005</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>Recurrent cutaneous angiosarcoma of the ear masquerading as atypical fibroxanthoma</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Gonzalez</surname><given-names>Cristian D.</given-names></name><degrees>BS, BA</degrees><xref rid="aff1" ref-type="aff">a</xref></contrib><contrib contrib-type="author"><name><surname>Hawkes</surname><given-names>Jason E.</given-names></name><degrees>MD</degrees><xref rid="aff1" ref-type="aff">a</xref></contrib><contrib contrib-type="author"><name><surname>Bowles</surname><given-names>Tawnya L.</given-names></name><degrees>MD, FACS</degrees><email>tawnya.bowles@imail.org</email><xref rid="aff2" ref-type="aff">b</xref><xref rid="aff3" ref-type="aff">c</xref><xref rid="cor1" ref-type="corresp">∗</xref></contrib></contrib-group><aff id="aff1"><label>a</label>Department of Dermatology, University of Utah School of Medicine, Salt Lake City, Utah</aff><aff id="aff2"><label>b</label>Melanoma and Cutaneous Oncology Program, Huntsman Cancer Institute, University of Utah, Salt Lake City, Utah</aff><aff id="aff3"><label>c</label>Intermountain Health Care, Salt Lake City, Utah</aff><author-notes><corresp id="cor1"><label>∗</label>Correspondence to: Tawnya L. Bowles, MD, FACS, Department of Surgery, Intermountain Medical Center, 5169 Cottonwood Street, Suite 440, Murray, UT 84107.Department of SurgeryIntermountain Medical Center5169 Cottonwood StreetSuite 440MurrayUT84107 <email>tawnya.bowles@imail.org</email></corresp></author-notes><pub-date pub-type="pmc-release"><day>05</day><month>12</month><year>2016</year></pub-date><pmc-comment> PMC Release delay is 0 months and 0 days and was based on .</pmc-comment>
<pub-date pub-type="collection"><month>11</month><year>2016</year></pub-date><pub-date pub-type="epub"><day>05</day><month>12</month><year>2016</year></pub-date><volume>2</volume><issue>6</issue><fpage>445</fpage><lpage>447</lpage><permissions><copyright-statement>© 2016 by the American Academy of Dermatology, Inc. Published by Elsevier, Inc.</copyright-statement><copyright-year>2016</copyright-year><copyright-holder>American Academy of Dermatology, Inc.</copyright-holder><license license-type="CC BY-NC-ND" xlink:href="http://creativecommons.org/licenses/by-nc-nd/4.0/"><license-p>This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).</license-p></license></permissions><kwd-group id="kwrds0010"><title>Key words</title><kwd>cutaneous angiosarcoma</kwd><kwd>external ear sarcoma</kwd><kwd>vascular tumor</kwd></kwd-group><kwd-group id="kwrds0015"><title>Abbreviations used</title><kwd>AFX, atypical fibroxanthoma</kwd><kwd>ERG, erythroblast transformation-specific–related gene</kwd></kwd-group></article-meta></front><body><sec id="sec1"><title>Introduction</title><p>Cutaneous angiosarcoma is a rare, malignant, vascular neoplasm of the skin commonly diagnosed in the head and neck regions. Involvement of the ear is rare.<xref rid="bib1" ref-type="bibr"><sup>1</sup></xref> Although no definitive surgical guidelines exist, wide resection with possible adjuvant chemoradiation is generally accepted as the standard treatment for resectable lesions.<xref rid="bib2" ref-type="bibr"><sup>2</sup></xref> We present the case of an elderly man with extensive angiosarcoma of the ear masquerading as atypical fibroxanthoma (AFX) necessitating total auriculectomy and combined radiation therapy. Our case also shows the utility of the erythroblast transformation-specific–related gene (ERG) staining for difficult-to-diagnose angiosarcomas.</p></sec><sec id="sec2"><title>Case report</title><p>A 65-year-old man presented for evaluation of a recurrent nodule of the left ear. Three years prior, he presented with a 6-mm papule of the left superior helix. Punch biopsy at the time found a spindle cell tumor with negative immunostaining for S-100, CD34, and MNF-116 (antipan cytokeratin antibody). He was subsequently referred to Mohs surgery for complete excision. One year later, he presented with a new 5-mm nodule at the same location on the ear. Punch biopsy at the time revealed an extensive residual dermal neoplasm composed of cytologically atypical and pleomorphic spindle cells. Immunostaining for S-100, CD34, and MNF-116 were negative, thus, favoring a diagnosis of recurrent AFX. Additional immunostaining was not done at that time, and the patient underwent radical resection of the left ear with 1-cm margins. The patient denied a history of radiation or trauma to the head and neck region. He denied past chemical exposure to arsenic, thorium dioxide, or vinyl chloride.</p><p>On examination, the patient had a 2.0- × 2.0-cm violaceous nodule on the superior helical rim of the left ear (<xref rid="fig1" ref-type="fig">Fig 1</xref>). A punch biopsy was sent for routine histologic evaluation and immunostaining, including ERG and CD31 staining. Hematoxylin-eosin stain showed a poorly circumscribed dermal neoplasm composed of atypical, pleomorphic spindle cells dissecting dermal collagen bundles (<xref rid="fig2" ref-type="fig">Fig 2</xref>, <italic>A</italic>). The neoplastic cells were positive for ERG (<xref rid="fig2" ref-type="fig">Fig 2</xref>, <italic>B</italic>) and CD31 (<xref rid="fig2" ref-type="fig">Fig 2</xref>, <italic>C</italic>). Immunostains for S-100, Melan-A, MNF-116, and CD34 were negative. The patient's initial surgical specimens were subsequently re-evaluated and found to be positive for ERG and CD31, thus, confirming a diagnosis of recurrent angiosarcoma. Computed tomographic imaging of the neck, chest, abdomen and pelvis showed no evidence of metastatic disease. A left total auriculectomy was performed to obtain the recommended 2-cm surgical margins. Postoperative histopathologic examination of the resected tissue found negative surgical margins. The patient was subsequently treated with adjuvant radiation therapy (60 Gy) given his high risk for local recurrence. The patient remains disease free with no signs of recurrence 9 months after treatment.</p></sec><sec id="sec3"><title>Discussion</title><p>Cutaneous angiosarcomas are rare, rapidly growing, malignant vascular neoplasms of vascular endothelial cells. Predisposing risk factors include postradiation therapy (eg, breast cancer therapy), vascular insufficiency, chronic lymphedema, trauma, and chemical exposure (eg, vinyl chloride, thorium dioxide, and arsenic). However, the exact etiology is unknown in most cases.</p><p>Angiosarcomas may present in a variety of ways (eg, purpuric/vascular, nodular, eczematous, rosacealike, and ulcerative) and may be difficult to diagnose in the early stages of development. Angiosarcomas are most commonly diagnosed in elderly Caucasians, and lesions typically involve the head and neck region.<xref rid="bib1" ref-type="bibr"><sup>1</sup></xref> Cutaneous involvement of the external ear is very rare.<xref rid="bib3" ref-type="bibr">3</xref>, <xref rid="bib4" ref-type="bibr">4</xref> Aggressive angiosarcoma involving the ear represents a challenging surgical scenario and may require extensive surgical resection to obtain negative tissue margins, as demonstrated in this case.</p><p>The diagnosis of angiosarcoma requires a high index of suspicion and, therefore, should be considered in the differential diagnosis for all vascular lesions on the head and neck of an elderly patient. Biopsy is necessary to differentiate between angiosarcoma and other benign or malignant neoplasms. For recurring lesions, an incisional biopsy is imperative. In a recent review by the French Sarcoma Group, more than 50% of angiosarcoma patients had evidence of widely metastatic disease at the time of diagnosis, frequently involving the lymph nodes, bone, and internal organs.<xref rid="bib5" ref-type="bibr"><sup>5</sup></xref> Therefore, computed tomography or positron emission tomography imaging is also crucial for determining the extent of disease before treatment.</p><p>The treatment of angiosarcoma can be challenging, and the prognosis associated with these neoplasms is poor.<xref rid="bib5" ref-type="bibr"><sup>5</sup></xref> Although no definitive surgical guidelines exist to guide the management of cutaneous disease, wide resection with possible adjuvant chemoradiation is preferred.<xref rid="bib2" ref-type="bibr"><sup>2</sup></xref> However, no randomized clinical trials or evidence-based guidelines are available for the treatment of angiosarcoma. Preclinical studies suggest a potential role for β-blockers in the treatment of angiosarcoma and identify phosphoinositide-dependent protein kinase 1 as a promising therapeutic target.<xref rid="bib6" ref-type="bibr">6</xref>, <xref rid="bib7" ref-type="bibr">7</xref></p><p>The differential diagnosis for angiosarcoma includes other spindle cell tumors, such as AFX, spindle cell and desmoplastic melanoma, squamous cell carcinoma, leiomyosarcoma, dermatofibrosarcoma protuberans, malignant fibrous histiocytoma, and myofibrosarcoma. Immunohistochemical studies are necessary to distinguish between these tumors.<xref rid="bib8" ref-type="bibr"><sup>8</sup></xref> Stains for S-100 and cytokeratins are negative for angiosarcoma and distinguish this tumor from a spindled cell melanoma and squamous cell, respectively. Positive immunostaining for CD31, CD34, factor VIII–related antigen, and ERG are frequently observed in angiosarcoma and should be performed when this diagnosis is suspected. AFX is considered a diagnosis of exclusion and should be considered after ruling out all other differential diagnoses.</p><p><italic>ERG</italic> is a proto-oncogene in the erythroblast transformation specific transcription factor family that is expressed in endothelial cells and appears to regulate angiogenesis.<xref rid="bib9" ref-type="bibr"><sup>9</sup></xref> Positive staining for CD31 and ERG are more sensitive markers for angiosarcoma than CD34.<xref rid="bib10" ref-type="bibr"><sup>10</sup></xref> ERG staining may provide additional diagnostic utility for atypical vascular tumors with equivocal staining for CD31 or CD34 as seen in this case. However, ERG staining has limitations, including the lack of large validation studies, weak staining patterns in one-quarter of angiosarcoma cases, and positive staining in other benign and malignant neoplasms (eg, hemangiomas, prostatic angiosarcoma, and Ewing's sarcoma).<xref rid="bib10" ref-type="bibr"><sup>10</sup></xref></p><p>We present the case of an elderly man with extensive angiosarcoma of the ear masquerading as AFX. This report highlights the diagnostic and surgical challenges associated with spindle cell neoplasms of the skin and ear. 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A large erythematous, violaceous nodule on the superior helical rim of the left ear.</p></caption><graphic xlink:href="gr1"></graphic></fig><fig id="fig2"><label>Fig 2</label><caption><p>Histologic images. <bold>A</bold>, Hematoxylin-eosin stain; <bold>B</bold>, ERG stain; <bold>C</bold>, CD31 stain; original magnifications: <bold>A</bold> through <bold>C</bold>, ×100.</p></caption><graphic xlink:href="gr2"></graphic></fig></floats-group></pmc></record>Pour manipuler ce document sous Unix (Dilib)
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