ELEPHANTIASIS NOSTRAS VERRUCOSA: SUCCESSFUL SURGICAL TREATMENT IN A GROSS DEFORMITY
Identifieur interne : 002557 ( Pmc/Corpus ); précédent : 002556; suivant : 002558ELEPHANTIASIS NOSTRAS VERRUCOSA: SUCCESSFUL SURGICAL TREATMENT IN A GROSS DEFORMITY
Auteurs : Purificaci N Gacto-Sánchez ; P. Fernandez-Ortega ; Jj Pereyra-RodríguezSource :
- Indian Journal of Dermatology [ 0019-5154 ] ; 2009.
Url:
DOI: 10.4103/0019-5154.53178
PubMed: 20101321
PubMed Central: 2807165
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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">ELEPHANTIASIS NOSTRAS VERRUCOSA: SUCCESSFUL SURGICAL TREATMENT IN A GROSS DEFORMITY</title><author><name sortKey="Gacto Sanchez, Purificaci N" sort="Gacto Sanchez, Purificaci N" uniqKey="Gacto Sanchez P" first="Purificaci N" last="Gacto-Sánchez">Purificaci N Gacto-Sánchez</name><affiliation><nlm:aff id="AF0001"></nlm:aff></affiliation></author><author><name sortKey="Fernandez Ortega, P" sort="Fernandez Ortega, P" uniqKey="Fernandez Ortega P" first="P" last="Fernandez-Ortega">P. Fernandez-Ortega</name><affiliation><nlm:aff id="AF0001"></nlm:aff></affiliation></author><author><name sortKey="Pereyra Rodriguez, Jj" sort="Pereyra Rodriguez, Jj" uniqKey="Pereyra Rodriguez J" first="Jj" last="Pereyra-Rodríguez">Jj Pereyra-Rodríguez</name><affiliation><nlm:aff id="AF0002"><italic>From the Department of Dermatology, Virgen del Rocío University Hospitals, Sevilla, Spain. E-mail:<email xlink:href="purigacto@gmail.com">purigacto@gmail.com</email></italic></nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">20101321</idno><idno type="pmc">2807165</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC2807165</idno><idno type="RBID">PMC:2807165</idno><idno type="doi">10.4103/0019-5154.53178</idno><date when="2009">2009</date><idno type="wicri:Area/Pmc/Corpus">002557</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">002557</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">ELEPHANTIASIS NOSTRAS VERRUCOSA: SUCCESSFUL SURGICAL TREATMENT IN A GROSS DEFORMITY</title><author><name sortKey="Gacto Sanchez, Purificaci N" sort="Gacto Sanchez, Purificaci N" uniqKey="Gacto Sanchez P" first="Purificaci N" last="Gacto-Sánchez">Purificaci N Gacto-Sánchez</name><affiliation><nlm:aff id="AF0001"></nlm:aff></affiliation></author><author><name sortKey="Fernandez Ortega, P" sort="Fernandez Ortega, P" uniqKey="Fernandez Ortega P" first="P" last="Fernandez-Ortega">P. Fernandez-Ortega</name><affiliation><nlm:aff id="AF0001"></nlm:aff></affiliation></author><author><name sortKey="Pereyra Rodriguez, Jj" sort="Pereyra Rodriguez, Jj" uniqKey="Pereyra Rodriguez J" first="Jj" last="Pereyra-Rodríguez">Jj Pereyra-Rodríguez</name><affiliation><nlm:aff id="AF0002"><italic>From the Department of Dermatology, Virgen del Rocío University Hospitals, Sevilla, Spain. E-mail:<email xlink:href="purigacto@gmail.com">purigacto@gmail.com</email></italic></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="2009">2009</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Sisto, K" uniqKey="Sisto K">K Sisto</name></author><author><name sortKey="Khachemoune, A" uniqKey="Khachemoune A">A Khachemoune</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Duckworth, Al" uniqKey="Duckworth A">AL Duckworth</name></author><author><name sortKey="Husain, J" uniqKey="Husain J">J Husain</name></author><author><name sortKey="Deheer, P" uniqKey="Deheer P">P Deheer</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Iwao, F" uniqKey="Iwao F">F Iwao</name></author><author><name sortKey="Sato Matsumura, Kc" uniqKey="Sato Matsumura K">KC Sato-Matsumura</name></author><author><name sortKey="Sawamura, D" uniqKey="Sawamura D">D Sawamura</name></author><author><name sortKey="Shimizu, H" uniqKey="Shimizu H">H Shimizu</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Zouboulis, Cc" uniqKey="Zouboulis C">CC Zouboulis</name></author><author><name sortKey="Bicz, S" uniqKey="Bicz S">S Biczó</name></author><author><name sortKey="Gollnick, H" uniqKey="Gollnick H">H Gollnick</name></author><author><name sortKey="Reupke, Hj" uniqKey="Reupke H">HJ Reupke</name></author><author><name sortKey="Rinck, G" uniqKey="Rinck G">G Rinck</name></author><author><name sortKey="Szab, M" uniqKey="Szab M">M Szabó</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Boyd, J" uniqKey="Boyd J">J Boyd</name></author><author><name sortKey="Sloan, S" uniqKey="Sloan S">S Sloan</name></author><author><name sortKey="Meffert, J" uniqKey="Meffert J">J Meffert</name></author></analytic></biblStruct></listBibl></div1></back></TEI><pmc article-type="letter"><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="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</publisher-name><publisher-loc>India</publisher-loc></publisher></journal-meta><article-meta><article-id pub-id-type="pmid">20101321</article-id><article-id pub-id-type="pmc">2807165</article-id><article-id pub-id-type="publisher-id">IJD-54-189</article-id><article-id pub-id-type="doi">10.4103/0019-5154.53178</article-id><article-categories><subj-group subj-group-type="heading"><subject>Correspondence</subject></subj-group></article-categories><title-group><article-title>ELEPHANTIASIS NOSTRAS VERRUCOSA: SUCCESSFUL SURGICAL TREATMENT IN A GROSS DEFORMITY</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Gacto-Sánchez</surname><given-names>Purificación</given-names></name><xref ref-type="aff" rid="AF0001"></xref></contrib><contrib contrib-type="author"><name><surname>Fernandez-Ortega</surname><given-names>P</given-names></name><xref ref-type="aff" rid="AF0001"></xref></contrib><contrib contrib-type="author"><name><surname>Pereyra-Rodríguez</surname><given-names>JJ</given-names></name><xref ref-type="aff" rid="AF0002">1</xref></contrib></contrib-group><aff id="AF0001"><italic>From the Departments of Plastic and Reconstructive Surgery, Virgen del Rocío University Hospitals, Sevilla, Spain</italic></aff><aff id="AF0002"><label>1</label><italic>From the Department of Dermatology, Virgen del Rocío University Hospitals, Sevilla, Spain. E-mail:<email xlink:href="purigacto@gmail.com">purigacto@gmail.com</email></italic></aff><pub-date pub-type="ppub"><season>Apr-Jun</season><year>2009</year></pub-date><volume>54</volume><issue>2</issue><fpage>189</fpage><lpage>190</lpage><permissions><copyright-statement>© Indian Journal of Dermatology</copyright-statement><copyright-year>2009</copyright-year><license license-type="open-access" xlink:href="http://creativecommons.org/licenses/by/2.0/"><license-p>This is an open-access article distributed under the terms of 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></article-meta></front><body><p>Sir,</p><p>Elephantiasis nostras verrucosa (ENV) is a rare, chronic, deforming disorder characterized by hyperkeratosis and papillomatosis of the epidermis with underlying woody fibrosis of the dermis and subcutaneous tissue. No standard treatment for this rare cutaneous manifestation is available.[<xref ref-type="bibr" rid="CIT1">1</xref>] In this case report, we describe surgical treatment that is proved to be helpful when performed in addition to physiotherapy.</p><p>A 51-year old male was presented to our clinic with a 15-year history of a pendulum-like mass arising from the anterior abdominal wall in its lower part covering penis and scrotal region [<xref ref-type="fig" rid="F0001">Figure 1</xref>]. The developed gross deformity was causing significant psychological alterations as well as social isolation to the patient. His medical history showed significant hyperlipidemia, obesity, hypertension, and diabetes mellitus. There was no personal history of filariasis or Milroy's disease in his family.</p><fig id="F0001" position="float"><label>Figure 1</label><caption><p>Pendulum-like mass arising from the anterior abdominal wall in its lower part covering penis and scrotal region</p></caption><graphic xlink:href="IJD-54-189-g001"></graphic></fig><p>Physical examination revealed generalized thickening and lichenification of the skin as well as multiple fistulas. No lymph nodes were palpable due to the woody nature of the tissue. Rectal examination revealed a slightly enlarged prostate without any palpable masses. Test results were negative for fecal occult blood. The patient underwent surgery to remove the mass. A compression stocking was used to treat the lymphedema. Ten months after the operation, we saw no signs of disease recurrence. The result was also aesthetically satisfactory [<xref ref-type="fig" rid="F0002">Figure 2</xref>].</p><fig id="F0002" position="float"><label>Figure 2</label><caption><p>Penis and scrotal region 6 months after surgical treatment</p></caption><graphic xlink:href="IJD-54-189-g002"></graphic></fig><p>Biopsy specimens revealed pseudoepitheliomatous hyperplasia with dilated lymphatic spaces in the dermis, accompanied by chronic inflammation and fibroblast proliferation. No evidence of neoplastic change was observed.</p><p>Elephantiasis is a clinical description of distinct cutaneous changes associated with a chronic underlying dysfunction in the lymphatic system. Many factors may lead to chronic lymphatic obstruction and stasis.[<xref ref-type="bibr" rid="CIT1">1</xref>] The common mechanism is an underlying lymphatic obstruction leading to impaired lymphatic drainage with abnormal accumulation of interstitial fluid and subsequent development of lymphedema. The overlying epidermis slowly develops a cobblestoned, verrucous appearance.[<xref ref-type="bibr" rid="CIT1">1</xref><xref ref-type="bibr" rid="CIT2">2</xref>] The precise role of some pathogenic organism, either bacterial or filarial, in “idiopathic” lymphatic obstruction is uncertain. Streptococcal and, less often, staphylococcal lymphangitis is considered to be the most likely causative agent.[<xref ref-type="bibr" rid="CIT1">1</xref><xref ref-type="bibr" rid="CIT2">2</xref>]</p><p>ENV and papillomatosis cutis carcinoides share many features. The latter can morphologically resemble ENV with single or multiple verrucous, partially ulcerated tumors on the chronically irritated edematous extremity. Both are histologically characterized by pseudoepitheliomatous hyperplasia. The diagnosis of papillomatosis cutis carcinoides is established by biopsy.[<xref ref-type="bibr" rid="CIT3">3</xref>]</p><p>Therapeutic efforts should aim to reduce lymph stasis, which will also improve the cutaneous changes. The mainstay of therapy remains prophylactic antibiotics, compression, and elevation of the affected limb.[<xref ref-type="bibr" rid="CIT3">3</xref>] Systemic retinoids, specifically etretinate, are useful in the treatment of diseases that are characterized by epidermal proliferation, as well as disorders of keratinization.[<xref ref-type="bibr" rid="CIT4">4</xref>] The value of topically applied tazarotene gel with its potent retinoid profile and low potential for systemic effects has still to be determined.[<xref ref-type="bibr" rid="CIT5">5</xref>] Our patient had a history of hyperlipidemia and refused systemic treatments. Hence, systemic retinoid therapy was deemed inappropriate.</p><p>ENV should be included in the differential diagnosis of chronic lymphedema and be differentiated from papillomatosis cutis carcinoides. Early diagnosis is paramount. Without appropriate early intervention, ENV continues to worsen and may result in gross deformity requiring amputation.[<xref ref-type="bibr" rid="CIT1">1</xref><xref ref-type="bibr" rid="CIT3">3</xref>]</p></body><back><ref-list><title>References</title><ref id="CIT1"><label>1</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Sisto</surname><given-names>K</given-names></name><name><surname>Khachemoune</surname><given-names>A</given-names></name></person-group><article-title>Elephantiasis nostras verrucosa: A review</article-title><source>Am J Clin Dermatol</source><year>2008</year><volume>9</volume><fpage>141</fpage><lpage>6</lpage><pub-id pub-id-type="pmid">18429642</pub-id></element-citation></ref><ref id="CIT2"><label>2</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Duckworth</surname><given-names>AL</given-names></name><name><surname>Husain</surname><given-names>J</given-names></name><name><surname>Deheer</surname><given-names>P</given-names></name></person-group><article-title>Elephantiasis nostras verrucosa or “mossy foot lesions” in lymphedema praecox: Report of a case</article-title><source>J Am Podiatr Med Assoc</source><year>2008</year><volume>98</volume><fpage>66</fpage><lpage>9</lpage><pub-id pub-id-type="pmid">18202337</pub-id></element-citation></ref><ref id="CIT3"><label>3</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Iwao</surname><given-names>F</given-names></name><name><surname>Sato-Matsumura</surname><given-names>KC</given-names></name><name><surname>Sawamura</surname><given-names>D</given-names></name><name><surname>Shimizu</surname><given-names>H</given-names></name></person-group><article-title>Elephantiasis nostras verrucosa successfully treated by surgical debridement</article-title><source>Dermatol Surg</source><year>2004</year><volume>30</volume><fpage>939</fpage><lpage>41</lpage><pub-id pub-id-type="pmid">15171776</pub-id></element-citation></ref><ref id="CIT4"><label>4</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Zouboulis</surname><given-names>CC</given-names></name><name><surname>Biczó</surname><given-names>S</given-names></name><name><surname>Gollnick</surname><given-names>H</given-names></name><name><surname>Reupke</surname><given-names>HJ</given-names></name><name><surname>Rinck</surname><given-names>G</given-names></name><name><surname>Szabó</surname><given-names>M</given-names></name><etal></etal></person-group><article-title>Elephantiasis nostras verrucosa: beneficial effect of oral etretinate therapy</article-title><source>Br J Dermatol</source><year>1992</year><volume>127</volume><fpage>411</fpage><lpage>6</lpage><pub-id pub-id-type="pmid">1419764</pub-id></element-citation></ref><ref id="CIT5"><label>5</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Boyd</surname><given-names>J</given-names></name><name><surname>Sloan</surname><given-names>S</given-names></name><name><surname>Meffert</surname><given-names>J</given-names></name></person-group><article-title>Elephantiasis nostrum verrucosa of the abdomen: Clinical results with tazarotene</article-title><source>J Drugs Dermatol</source><year>2004</year><volume>3</volume><fpage>446</fpage><lpage>8</lpage><pub-id pub-id-type="pmid">15303792</pub-id></element-citation></ref></ref-list></back></pmc></record>Pour manipuler ce document sous Unix (Dilib)
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