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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Detection of Recurrent Cutaneous Angiosarcoma of Lower Extremity with <sup>18</sup>
F-Fluorodeoxyglucose Positron Emission Tomography-Computed Tomography: Report of Three Cases</title>
<author><name sortKey="Sharma, Punit" sort="Sharma, Punit" uniqKey="Sharma P" first="Punit" last="Sharma">Punit Sharma</name>
<affiliation><nlm:aff id="aff1"></nlm:aff>
</affiliation>
</author>
<author><name sortKey="Singh, Harmandeep" sort="Singh, Harmandeep" uniqKey="Singh H" first="Harmandeep" last="Singh">Harmandeep Singh</name>
<affiliation><nlm:aff id="aff1"></nlm:aff>
</affiliation>
</author>
<author><name sortKey="Singhal, Abhinav" sort="Singhal, Abhinav" uniqKey="Singhal A" first="Abhinav" last="Singhal">Abhinav Singhal</name>
<affiliation><nlm:aff id="aff1"></nlm:aff>
</affiliation>
</author>
<author><name sortKey="Bal, Chandrasekhar" sort="Bal, Chandrasekhar" uniqKey="Bal C" first="Chandrasekhar" last="Bal">Chandrasekhar Bal</name>
<affiliation><nlm:aff id="aff1"></nlm:aff>
</affiliation>
</author>
<author><name sortKey="Kumar, Rakesh" sort="Kumar, Rakesh" uniqKey="Kumar R" first="Rakesh" last="Kumar">Rakesh Kumar</name>
<affiliation><nlm:aff id="aff1"></nlm:aff>
</affiliation>
</author>
</titleStmt>
<publicationStmt><idno type="wicri:source">PMC</idno>
<idno type="pmid">23723498</idno>
<idno type="pmc">3667310</idno>
<idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3667310</idno>
<idno type="RBID">PMC:3667310</idno>
<idno type="doi">10.4103/0019-5154.110859</idno>
<date when="2013">2013</date>
<idno type="wicri:Area/Pmc/Corpus">002225</idno>
<idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">002225</idno>
</publicationStmt>
<sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Detection of Recurrent Cutaneous Angiosarcoma of Lower Extremity with <sup>18</sup>
F-Fluorodeoxyglucose Positron Emission Tomography-Computed Tomography: Report of Three Cases</title>
<author><name sortKey="Sharma, Punit" sort="Sharma, Punit" uniqKey="Sharma P" first="Punit" last="Sharma">Punit Sharma</name>
<affiliation><nlm:aff id="aff1"></nlm:aff>
</affiliation>
</author>
<author><name sortKey="Singh, Harmandeep" sort="Singh, Harmandeep" uniqKey="Singh H" first="Harmandeep" last="Singh">Harmandeep Singh</name>
<affiliation><nlm:aff id="aff1"></nlm:aff>
</affiliation>
</author>
<author><name sortKey="Singhal, Abhinav" sort="Singhal, Abhinav" uniqKey="Singhal A" first="Abhinav" last="Singhal">Abhinav Singhal</name>
<affiliation><nlm:aff id="aff1"></nlm:aff>
</affiliation>
</author>
<author><name sortKey="Bal, Chandrasekhar" sort="Bal, Chandrasekhar" uniqKey="Bal C" first="Chandrasekhar" last="Bal">Chandrasekhar Bal</name>
<affiliation><nlm:aff id="aff1"></nlm:aff>
</affiliation>
</author>
<author><name sortKey="Kumar, Rakesh" sort="Kumar, Rakesh" uniqKey="Kumar R" first="Rakesh" last="Kumar">Rakesh Kumar</name>
<affiliation><nlm:aff id="aff1"></nlm:aff>
</affiliation>
</author>
</analytic>
<series><title level="j">Indian Journal of Dermatology</title>
<idno type="ISSN">0019-5154</idno>
<idno type="eISSN">1998-3611</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>Cutaneous angiosarcomas (CAS) are uncommon, aggressive tumours. Very rarely, they arise from the lower extremity. Such tumours are usually associated with chronic lymphedema, a phenomenon known as Stewart-Treves Syndrome. Treatment is usually radical surgery with adjuvant therapy (radiotherapy/chemotherapy). Recurrence rate after primary treatment is high. Because of post therapy changes, conventional imaging has limited specificity for diagnosing recurrence. <sup>18</sup>
F-Fluorodeoxyglucose (<sup>18</sup>
F-FDG) positron emission tomography-computed tomography (PET-CT) might be useful in such patients. It can demonstrate local recurrence along with distant metastasis, if any and can have significant impact on patient management. We here present three cases of recurrent CAS of lower extremity diagnosed with <sup>18</sup>
F-FDG PET-CT.</p>
</div>
</front>
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</TEI>
<pmc article-type="case-report"><pmc-dir>properties open_access</pmc-dir>
<front><journal-meta><journal-id journal-id-type="nlm-ta">Indian J Dermatol</journal-id>
<journal-id journal-id-type="iso-abbrev">Indian J Dermatol</journal-id>
<journal-id journal-id-type="publisher-id">IJD</journal-id>
<journal-title-group><journal-title>Indian Journal of Dermatology</journal-title>
</journal-title-group>
<issn pub-type="ppub">0019-5154</issn>
<issn pub-type="epub">1998-3611</issn>
<publisher><publisher-name>Medknow Publications & Media Pvt Ltd</publisher-name>
<publisher-loc>India</publisher-loc>
</publisher>
</journal-meta>
<article-meta><article-id pub-id-type="pmid">23723498</article-id>
<article-id pub-id-type="pmc">3667310</article-id>
<article-id pub-id-type="publisher-id">IJD-58-242d</article-id>
<article-id pub-id-type="doi">10.4103/0019-5154.110859</article-id>
<article-categories><subj-group subj-group-type="heading"><subject>E-Case Report</subject>
</subj-group>
</article-categories>
<title-group><article-title>Detection of Recurrent Cutaneous Angiosarcoma of Lower Extremity with <sup>18</sup>
F-Fluorodeoxyglucose Positron Emission Tomography-Computed Tomography: Report of Three Cases</article-title>
</title-group>
<contrib-group><contrib contrib-type="author"><name><surname>Sharma</surname>
<given-names>Punit</given-names>
</name>
<xref ref-type="aff" rid="aff1"></xref>
</contrib>
<contrib contrib-type="author"><name><surname>Singh</surname>
<given-names>Harmandeep</given-names>
</name>
<xref ref-type="aff" rid="aff1"></xref>
</contrib>
<contrib contrib-type="author"><name><surname>Singhal</surname>
<given-names>Abhinav</given-names>
</name>
<xref ref-type="aff" rid="aff1"></xref>
</contrib>
<contrib contrib-type="author"><name><surname>Bal</surname>
<given-names>Chandrasekhar</given-names>
</name>
<xref ref-type="aff" rid="aff1"></xref>
</contrib>
<contrib contrib-type="author"><name><surname>Kumar</surname>
<given-names>Rakesh</given-names>
</name>
<xref ref-type="aff" rid="aff1"></xref>
<xref ref-type="corresp" rid="cor1"></xref>
</contrib>
</contrib-group>
<aff id="aff1"><italic>From the Department of Nuclear Medicine, All India Institute of Medical Sciences, New Delhi, India</italic>
</aff>
<author-notes><corresp id="cor1"><italic><bold>Address for correspondence:</bold>
Dr. Rakesh Kumar, E-81, Ansari Nagar (East), AIIMS Campus, New Delhi - 110 029, India. E-mail: <email xlink:href="rkphulia@yahoo.com">rkphulia@yahoo.com</email>
</italic>
</corresp>
</author-notes>
<pub-date pub-type="ppub"><season>May-Jun</season>
<year>2013</year>
</pub-date>
<volume>58</volume>
<issue>3</issue>
<fpage seq="d">242</fpage>
<lpage>242</lpage>
<history><date date-type="received"><month>3</month>
<year>2012</year>
</date>
<date date-type="accepted"><month>4</month>
<year>2012</year>
</date>
</history>
<permissions><copyright-statement>Copyright: © Indian Journal of Dermatology</copyright-statement>
<copyright-year>2013</copyright-year>
<license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by-nc-sa/3.0"><license-p>This is an open-access article distributed under the terms of the Creative Commons Attribution-Noncommercial-Share Alike 3.0 Unported, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.</license-p>
</license>
</permissions>
<abstract><p>Cutaneous angiosarcomas (CAS) are uncommon, aggressive tumours. Very rarely, they arise from the lower extremity. Such tumours are usually associated with chronic lymphedema, a phenomenon known as Stewart-Treves Syndrome. Treatment is usually radical surgery with adjuvant therapy (radiotherapy/chemotherapy). Recurrence rate after primary treatment is high. Because of post therapy changes, conventional imaging has limited specificity for diagnosing recurrence. <sup>18</sup>
F-Fluorodeoxyglucose (<sup>18</sup>
F-FDG) positron emission tomography-computed tomography (PET-CT) might be useful in such patients. It can demonstrate local recurrence along with distant metastasis, if any and can have significant impact on patient management. We here present three cases of recurrent CAS of lower extremity diagnosed with <sup>18</sup>
F-FDG PET-CT.</p>
</abstract>
<kwd-group><kwd><italic>Angiosarcoma</italic>
</kwd>
<kwd><italic>cutaneous</italic>
</kwd>
<kwd><italic>lower extremity</italic>
</kwd>
<kwd><italic>positron emission tomography-computed tomography</italic>
</kwd>
<kwd><italic>recurrence</italic>
</kwd>
</kwd-group>
</article-meta>
</front>
<body><sec id="sec1-1"><title>Introduction</title>
<boxed-text position="float"><p><bold>What was known?</bold>
</p>
<p>1. Cutaneous angiosarcomas of lower extremity are rare aggressive neoplasm.</p>
<p>2. Clinical examination or conventional imaging has limited specificity for diagnosing local recurrence after treatment of such tumours.</p>
</boxed-text>
<p>Cutaneous angiosarcomas (CAS) are rare tumours mostly seen in scalp of elderly patients.[<xref ref-type="bibr" rid="ref1">1</xref>
] CAS of lower extremity is rarer and usually seen in association with chronic lymphedema, the entity being known as Stewart-Treves Syndrome (STS).[<xref ref-type="bibr" rid="ref2">2</xref>
] These tumours are aggressive and have high rates of recurrence and metastasis.[<xref ref-type="bibr" rid="ref3">3</xref>
] As the primary tumours are treated with surgery, sometimes with adjuvant radiotherapy, it is often difficult to differentiate recurrent tumours from post therapy changes.[<xref ref-type="bibr" rid="ref3">3</xref>
] Metabolic imaging with <sup>18</sup>
F-Fluorodeoxyglucose (<sup>18</sup>
F-FDG) positron emission tomography-computed tomography (PET-CT) might be useful in such patients. We present here three cases of recurrent CAS of lower extremity detected with <sup>18</sup>
F-FDG PET-CT and briefly review the role it can play in management of such tumours.</p>
</sec>
<sec id="sec1-2"><title>Case Reports</title>
<sec id="sec2-1"><title>Case 1</title>
<p>A 39 year old female with history of chronic lymphedema of left leg, developed a non healing ulcer. Biopsy from the ulcer revealed angiosarcoma (STS). She underwent <sup>18</sup>
F-FDG PET-CT for staging. It revealed multiple <sup>18</sup>
F-FDG avid (SUVmax-4.5) cutaneous nodules on anterior aspect of left leg [Figures <xref ref-type="fig" rid="F1">1a</xref>
and <xref ref-type="fig" rid="F2">2a</xref>
-<xref ref-type="fig" rid="F2">c</xref>
] but no distant metastasis. She underwent radical surgery followed by four cycles of chemotherapy. <sup>18</sup>
F-FDG PET-CT was repeated to assess response one month after completion of chemotherapy. Complete resolution of cutaneous lesions was seen, suggesting complete response [Figures <xref ref-type="fig" rid="F1">1b</xref>
and <xref ref-type="fig" rid="F2">2d</xref>
-<xref ref-type="fig" rid="F2">f</xref>
]. The patient then lost to follow up and presented 2 years later with multiple cutaneous nodules in left leg. <sup>18</sup>
F-FDG PET-CT study was repeated for any distant metastasis. Multiple large <sup>18</sup>
F-FDG avid (SUVmax-7.3) necrotic cutaneous nodules were seen in left leg suggesting extensive recurrent disease [Figures <xref ref-type="fig" rid="F1">1c</xref>
and <xref ref-type="fig" rid="F2">2g</xref>
-<xref ref-type="fig" rid="F2">i</xref>
] but no distant metastasis was seen. The patient was advised for an amputation, which she refused. She is on palliative radio-chemotherapy.</p>
<fig id="F1" position="float"><label>Figure 1</label>
<caption><p>A 39 year old female with Stewart-Treves Syndrome (Case 1). Maximum intensity projection (MIP) PET images at baseline (a) reveal multiple <sup>18</sup>
F-FDG avid lesions in the left leg anteriorly. Repeat study done after surgery and chemotherapy (b) shows no such focus, except for <sup>18</sup>
F-FDG uptake in muscle (arrowhead). After 2 years a third PET-CT (c) was done, which show multiple <sup>18</sup>
F-FDG avid lesions in leg, extending upto left thigh suggesting recurrent disease</p>
</caption>
<graphic xlink:href="IJD-58-242d-g001"></graphic>
</fig>
<fig id="F2" position="float"><label>Figure 2</label>
<caption><p>PET-CT images of the above patient (Case 1). Upper row show baseline CT (a) PET (b) and PET-CT (c) images. The images show cutaneous and subcutaneous thickening (<italic>arrow</italic>
) with increased FDG uptake. PET-CT was done after surgery and chemotherapy (d-f) shows postoperative change with no significant <sup>18</sup>
F-FDG uptake suggesting complete response to therapy. Repeat PET-CT images (g-i) done after 2 years shows large necrotic FDG avid lesions in the left leg (arrow) suggesting recurrent disease. This was confirmed on biopsy</p>
</caption>
<graphic xlink:href="IJD-58-242d-g002"></graphic>
</fig>
</sec>
<sec id="sec2-2"><title>Case 2</title>
<p>A 58 year old woman with history of chronic lymphedema of left leg developed a 2.5 × 3 cm reddish patch over the left leg. Biopsy revealed angiosarcoma (STS). She underwent wide local excision of the tumour followed by skin grafting. Three months after the surgery, she complained of discomfort at the surgery site along with bone pains. Magnetic resonance imaging (MRI) of the lower limbs was suggestive of post-operative changes. Restaging <sup>18</sup>
F-FDG PET-CT was done to look for distant metastasis. No distant metastasis was seen [<xref ref-type="fig" rid="F3">Figure 3a</xref>
]. However, it revealed cutaneous thickening in left leg antero-laterally with increased <sup>18</sup>
F-FDG uptake (SUVmax-2.7) [<xref ref-type="fig" rid="F3">Figure 3b</xref>
-<xref ref-type="fig" rid="F3">e</xref>
], suspicious for local recurrence. The diagnosis was confirmed with biopsy. She underwent re-operation followed by adjuvant chemotherapy and is well at 6 month follow up.</p>
<fig id="F3" position="float"><label>Figure 3</label>
<caption><p>A 58 year old female with Stewart-Treves Syndrome (Case 2). She underwent PET-CT for suspected recurrence of cutaneous angiosarcoma of left lower limb. Whole body maximum intensity projection (MIP) PET image (a) does not show any distant metastasis. MIP PET images of lower limbs (b) reveal focally increased <sup>18</sup>
F-FDG uptake in the antero-lateral part of left leg (<italic>arrow</italic>
). Transaxial CT (c) PET (d) and PET-CT (e) images show cutaneous thickening with increased <sup>18</sup>
F-FDG uptake in antero-lateral part of left lower leg (<italic>arrow</italic>
), suspicious for local recurrence. This was confirmed with biopsy</p>
</caption>
<graphic xlink:href="IJD-58-242d-g003"></graphic>
</fig>
</sec>
<sec id="sec2-3"><title>Case 3</title>
<p>A 72 year old male developed 5 × 3.2 cm purplish-red cutaneous papule in the posterior aspect of left lower limb. There was no lymphedema. The tumour was resected and histopathology revealed angiosarcoma. Staging investigations at the time of surgery were negative. The patient presented with stiffness and discomfort at the operated site one year later. <sup>18</sup>
F-FDG PET-CT was done. It revealed cutaneous thickening with increased <sup>18</sup>
F-FDG uptake (SUVmax-3.3) in the left popliteal region [<xref ref-type="fig" rid="F4">Figure 4b</xref>
-<xref ref-type="fig" rid="F4">d</xref>
] but no distant metastasis [<xref ref-type="fig" rid="F4">Figure 4a</xref>
]. <sup>18</sup>
F-FDG PET-CT was suggestive of local recurrence only. The patient underwent surgery for removal of the tumour and is doing fine at 18 months follow up.</p>
<fig id="F4" position="float"><label>Figure 4</label>
<caption><p>A 72 year old male with cutaneous angiosarcoma of left lower limb (Case 3). The lesion was surgically resected. <sup>18</sup>
F-FDG PET-CT was done 1 year later for suspected local recurrence. Whole body maximum intensity projection (MIP) PET image (a) does not show any distant metastasis. Transaxial CT (b) PET (c) and PET-CT (d) images show <sup>18</sup>
F-FDG avid cutaneous thickening in the popliteal region of left leg extending to involve the underlying muscles (<italic>arrow</italic>
). Findings were suggestive of local recurrence and confirmed with biopsy</p>
</caption>
<graphic xlink:href="IJD-58-242d-g004"></graphic>
</fig>
</sec>
</sec>
<sec sec-type="discussion" id="sec1-3"><title>Discussion</title>
<p>CAS are high-grade and highly aggressive endothelial-cell tumours of vascular or lymphatic origin, with a high incidence of metastasis at presentation.[<xref ref-type="bibr" rid="ref3">3</xref>
–<xref ref-type="bibr" rid="ref5">5</xref>
] They constitute 5.4% of cutaneous soft tissue sarcomas.[<xref ref-type="bibr" rid="ref6">6</xref>
] It typically involves the head and neck, particularly the scalp. Angiosarcomas of the extremities are rare. Chronic lymphedema and previous radiotherapy are two well documented risk factor for development of CAS.[<xref ref-type="bibr" rid="ref2">2</xref>
<xref ref-type="bibr" rid="ref7">7</xref>
] Chronic lymphedema of any origin is associated with the development of angiosarcoma; a phenomenon known as STS.[<xref ref-type="bibr" rid="ref2">2</xref>
] The primary treatment is usually radical surgery. Despite radical surgery, recurrence rates are high. So, multimodality treatment (radiotherapy/chemotherapy) is usually employed and has been shown to be beneficial.[<xref ref-type="bibr" rid="ref3">3</xref>
] Even with multimodality treatment there is high risk of recurrence. Additional surgery in locally-recurrent disease to achieve a pathological complete resection may improve survival in such patients.[<xref ref-type="bibr" rid="ref8">8</xref>
] This highlights the need to diagnose recurrent disease at its earliest. Computed tomography (CT) and magnetic resonance imaging (MRI) are used for primary staging of these tumours, but their role is limited in recurrent tumours because of low specificity due to post surgical and post radiotherapy changes.</p>
<p><sup>18</sup>
F-FDG PET-CT is being utilised for wide array of tumours, including cutaneous neoplasms.[<xref ref-type="bibr" rid="ref9">9</xref>
] It has also been shown to be useful in primary angiosarcoma of various sites, even CAS.[<xref ref-type="bibr" rid="ref10">10</xref>
<xref ref-type="bibr" rid="ref11">11</xref>
] Vasanawala, <italic>et al</italic>
. previously reported the utility of <sup>18</sup>
F-FDG PET-CT for diagnosis and restaging in a case of CAS of scalp.[<xref ref-type="bibr" rid="ref12">12</xref>
] Similarly, Jensen, <italic>et al</italic>
. have shown the utility of <sup>18</sup>
F-FDG PET-CT in two cases of Stewart-Treves Syndrome.[<xref ref-type="bibr" rid="ref10">10</xref>
] Dawlatly, <italic>et al</italic>
. showed efficacy of <sup>18</sup>
F-FDG PET-CT in two cases of STS of lower extremity and found PET-CT to be helpful in demonstrating the extent of subcutaneous spread and planning surgical management.[<xref ref-type="bibr" rid="ref13">13</xref>
] However, to our knowledge utility of <sup>18</sup>
F-FDG PET-CT in recurrent CAS has not been reported till date. We found <sup>18</sup>
F-FDG PET-CT to be very useful. This is true for Stewart-Treves Syndrome (case 1 and 2) as well as spontaneous tumours (case 3). In all three patients <sup>18</sup>
F-FDG PET-CT was able to demonstrate recurrent disease. It could be superior to MRI for detecting local recurrence (case 2). The <sup>18</sup>
F-FDG avidity of recurrent tumour varied with SUVmax ranging from 2.7 to 7.3 and is probably related to biological aggressiveness. In case 1 the recurrent tumour was more aggressive as demonstrated by increase in SUVmax from baseline (4.5 to 7.3). Apart from demonstrating local recurrence another major utility of <sup>18</sup>
F-FDG PET-CT was to rule out distant metastasis at staging (case 1) or at restaging (case 2 and 3). This is especially important in these tumours given the high incidence metastasis.[<xref ref-type="bibr" rid="ref5">5</xref>
]</p>
<p>In conclusion, <sup>18</sup>
F-FDG PET-CT appears to be useful for diagnosing recurrent CAS. It influences patient management by demonstrating local recurrence and ruling in/out distant metastasis. These findings if confirmed in larger patient population might lead to integration of <sup>18</sup>
F-FDG PET-CT in management protocols of CAS.</p>
<boxed-text position="float"><p><bold>What is new?</bold>
</p>
<p>1. <sup>18</sup>
F-FDG PET-CT can be used for detecting local recurrence in cutaneous angiosarcoma of lower extremities.</p>
<p>2. It can also detect or rule out distant metastasis in this setting.</p>
</boxed-text>
</sec>
</body>
<back><fn-group><fn fn-type="supported-by"><p><bold>Source of Support:</bold>
Nil</p>
</fn>
<fn fn-type="conflict"><p><bold>Conflict of Interest:</bold>
Nil.</p>
</fn>
</fn-group>
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