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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">CHRONIC LINEAR ULCERATIONS OF THE INGUINO-CRURAL AND BUTTOCKS FOLDS</title><author><name sortKey="Khaled, Aida" sort="Khaled, Aida" uniqKey="Khaled A" first="Aida" last="Khaled">Aida Khaled</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Ezzine Sebai, Nadia" sort="Ezzine Sebai, Nadia" uniqKey="Ezzine Sebai N" first="Nadia" last="Ezzine-Sebai">Nadia Ezzine-Sebai</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Fazaa, Becima" sort="Fazaa, Becima" uniqKey="Fazaa B" first="Becima" last="Fazaa">Becima Fazaa</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Zeglaoui, Faten" sort="Zeglaoui, Faten" uniqKey="Zeglaoui F" first="Faten" last="Zeglaoui">Faten Zeglaoui</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Zermani, Rachida" sort="Zermani, Rachida" uniqKey="Zermani R" first="Rachida" last="Zermani">Rachida Zermani</name><affiliation><nlm:aff id="aff2"><italic>Department of pathology, Charles Nicolle Hospital – Tunis, Tunisia</italic></nlm:aff></affiliation></author><author><name sortKey="Kamoun, Mohamed Ridha" sort="Kamoun, Mohamed Ridha" uniqKey="Kamoun M" first="Mohamed Ridha" last="Kamoun">Mohamed Ridha Kamoun</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">21572806</idno><idno type="pmc">3088916</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3088916</idno><idno type="RBID">PMC:3088916</idno><idno type="doi">10.4103/0019-5154.77568</idno><date when="2011">2011</date><idno type="wicri:Area/Pmc/Corpus">002236</idno><idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">002236</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">CHRONIC LINEAR ULCERATIONS OF THE INGUINO-CRURAL AND BUTTOCKS FOLDS</title><author><name sortKey="Khaled, Aida" sort="Khaled, Aida" uniqKey="Khaled A" first="Aida" last="Khaled">Aida Khaled</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Ezzine Sebai, Nadia" sort="Ezzine Sebai, Nadia" uniqKey="Ezzine Sebai N" first="Nadia" last="Ezzine-Sebai">Nadia Ezzine-Sebai</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Fazaa, Becima" sort="Fazaa, Becima" uniqKey="Fazaa B" first="Becima" last="Fazaa">Becima Fazaa</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Zeglaoui, Faten" sort="Zeglaoui, Faten" uniqKey="Zeglaoui F" first="Faten" last="Zeglaoui">Faten Zeglaoui</name><affiliation><nlm:aff id="aff1"></nlm:aff></affiliation></author><author><name sortKey="Zermani, Rachida" sort="Zermani, Rachida" uniqKey="Zermani R" first="Rachida" last="Zermani">Rachida Zermani</name><affiliation><nlm:aff id="aff2"><italic>Department of pathology, Charles Nicolle Hospital – Tunis, Tunisia</italic></nlm:aff></affiliation></author><author><name sortKey="Kamoun, Mohamed Ridha" sort="Kamoun, Mohamed Ridha" uniqKey="Kamoun M" first="Mohamed Ridha" last="Kamoun">Mohamed Ridha Kamoun</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="2011">2011</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p>Vulvo-perineal Crohn's disease is a rare condition either when it is isolated or associated with digestive manifestations. In the former condition named metastatic Crohn's disease, it may constitute a diagnostic challenge and may be confused especially with other infectious or inflammatory disorders. We report a case of vulvo-perineal Crohn's disease in a 46-year-old woman. A 46-year-old woman was diagnosed with a vulvo-perineal Crohn's disease without digestive involvement. There was a chronic edema of the vulva with linear ulcerations on the inguino-crural regions and the buttocks fold, of 3 years. Treatment with metronidazole (1 g/day for 6 months) led to almost complete healing of the ulcerations with a sustained result. Physicians must be aware of the diverse manifestations and confusing presentations of vulvo-perineal Crohn's disease.</p></div></front><back><div1 type="bibliography"><listBibl><biblStruct><analytic><author><name sortKey="Guest, Gd" uniqKey="Guest G">GD Guest</name></author><author><name sortKey="Fink, Rl" uniqKey="Fink R">RL Fink</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Conscience, I" uniqKey="Conscience I">I Conscience</name></author><author><name sortKey="Perceau, G" uniqKey="Perceau G">G Perceau</name></author><author><name sortKey="Durlach, A" uniqKey="Durlach A">A Durlach</name></author><author><name sortKey="Bernard, P" uniqKey="Bernard P">P Bernard</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Kolivras, A" uniqKey="Kolivras A">A Kolivras</name></author><author><name sortKey="De Maubeuge, J" uniqKey="De Maubeuge J">J De Maubeuge</name></author><author><name sortKey="Andre, J" uniqKey="Andre J">J André</name></author><author><name sortKey="Song, M" uniqKey="Song M">M Song</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Preston, Pw" uniqKey="Preston P">PW Preston</name></author><author><name sortKey="Hudson, N" uniqKey="Hudson N">N Hudson</name></author><author><name sortKey="Lewis, Fm" uniqKey="Lewis F">FM Lewis</name></author></analytic></biblStruct><biblStruct><analytic><author><name sortKey="Bernstein, Lh" uniqKey="Bernstein L">LH Bernstein</name></author><author><name sortKey="Frank, Ms" uniqKey="Frank M">MS Frank</name></author><author><name sortKey="Brandt, Lj" uniqKey="Brandt L">LJ Brandt</name></author><author><name sortKey="Boley, Sj" uniqKey="Boley S">SJ Boley</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">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">21572806</article-id><article-id pub-id-type="pmc">3088916</article-id><article-id pub-id-type="publisher-id">IJD-56-101</article-id><article-id pub-id-type="doi">10.4103/0019-5154.77568</article-id><article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject></subj-group></article-categories><title-group><article-title>CHRONIC LINEAR ULCERATIONS OF THE INGUINO-CRURAL AND BUTTOCKS FOLDS</article-title></title-group><contrib-group><contrib contrib-type="author"><name><surname>Khaled</surname><given-names>Aida</given-names></name><xref ref-type="aff" rid="aff1"></xref><xref ref-type="corresp" rid="cor1"></xref></contrib><contrib contrib-type="author"><name><surname>Ezzine-Sebai</surname><given-names>Nadia</given-names></name><xref ref-type="aff" rid="aff1"></xref></contrib><contrib contrib-type="author"><name><surname>Fazaa</surname><given-names>Becima</given-names></name><xref ref-type="aff" rid="aff1"></xref></contrib><contrib contrib-type="author"><name><surname>Zeglaoui</surname><given-names>Faten</given-names></name><xref ref-type="aff" rid="aff1"></xref></contrib><contrib contrib-type="author"><name><surname>Zermani</surname><given-names>Rachida</given-names></name><xref ref-type="aff" rid="aff2">1</xref></contrib><contrib contrib-type="author"><name><surname>Kamoun</surname><given-names>Mohamed Ridha</given-names></name><xref ref-type="aff" rid="aff1"></xref></contrib></contrib-group><aff id="aff1"><italic>Department of Dermatology,Charles Nicolle Hospital – Tunis, Tunisia</italic></aff><aff id="aff2"><label>1</label><italic>Department of pathology, Charles Nicolle Hospital – Tunis, Tunisia</italic></aff><author-notes><corresp id="cor1"><italic><bold>Address for correspondence:</bold> Dr. Khaled Aida , Boulevard 9 Avril Tunis- 1006, Tunisia. E-mail: <email xlink:href="aida.khaled@rns.tn">aida.khaled@rns.tn</email></italic></corresp></author-notes><pub-date pub-type="ppub"><season>Jan-Feb</season><year>2011</year></pub-date><volume>56</volume><issue>1</issue><fpage>101</fpage><lpage>103</lpage><history><date date-type="received"><month>1</month><year>2009</year></date><date date-type="accepted"><month>7</month><year>2010</year></date></history><permissions><copyright-statement>Copyright: © Indian Journal of Dermatology</copyright-statement><copyright-year>2011</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>Vulvo-perineal Crohn's disease is a rare condition either when it is isolated or associated with digestive manifestations. In the former condition named metastatic Crohn's disease, it may constitute a diagnostic challenge and may be confused especially with other infectious or inflammatory disorders. We report a case of vulvo-perineal Crohn's disease in a 46-year-old woman. A 46-year-old woman was diagnosed with a vulvo-perineal Crohn's disease without digestive involvement. There was a chronic edema of the vulva with linear ulcerations on the inguino-crural regions and the buttocks fold, of 3 years. Treatment with metronidazole (1 g/day for 6 months) led to almost complete healing of the ulcerations with a sustained result. Physicians must be aware of the diverse manifestations and confusing presentations of vulvo-perineal Crohn's disease.</p></abstract><kwd-group><kwd><italic>Cutaneous Crohn's disease</italic></kwd><kwd><italic>vulvo-perineal Crohn's disease</italic></kwd><kwd><italic>metastatic Crohn's disease</italic></kwd></kwd-group></article-meta></front><body><sec sec-type="cases" id="sec1-1"><title>Case Report</title><p>A 46-year-old woman, having in her past medical history chronic juvenile arthritis with bilateral prosthetic hips, presented with painful vulvo-perineal ulcerations of 3 years. There was no diarrhea or recent weight loss. Cutaneous examination showed asymmetrical vulvar edema of the labia minora and labia majora with deep and linear ulcerations having verrucous borders located on the inguino-crural regions [<xref ref-type="fig" rid="F1">Figure 1</xref>] and the buttocks fold [<xref ref-type="fig" rid="F2">Figure 2</xref>]. On physical examination there was bilateral limited mobilization of the hips. A biopsy specimen was taken from the border of the vulvar ulceration. Histological examination showed under an epidermal hyperplasia [<xref ref-type="fig" rid="F3">Figure 3</xref>], an epithelioid granuloma of the dermis showing multinucleated giant cells without caseation [<xref ref-type="fig" rid="F4">Figure 4</xref>]. Direct immunofluorescence on cutaneous biopsy was negative. Biology and chest X-ray were normal. Koch's bacillus in sputum, microbiological studies (staining for micro-organisms and cultures) and tuberculin testing were negative.</p><fig id="F1" position="float"><label>Figure 1</label><caption><p>Asymmetrical vulvar edema with deep and linear ulcerations of the inguino-crural regions</p></caption><graphic xlink:href="IJD-56-101-g001"></graphic></fig><fig id="F2" position="float"><label>Figure 2</label><caption><p>Linear ulcerations with verrucous borders of the buttocks fold</p></caption><graphic xlink:href="IJD-56-101-g002"></graphic></fig><fig id="F3" position="float"><label>Figure 3</label><caption><p>Epidermal hyperplasia (H&E stain, 100×)</p></caption><graphic xlink:href="IJD-56-101-g003"></graphic></fig><fig id="F4" position="float"><label>Figure 4</label><caption><p>Epithelioid granuloma of the dermis showing multinucleated giant cells without caseation (H&E stain, 400×)</p></caption><graphic xlink:href="IJD-56-101-g004"></graphic></fig></sec><sec id="sec1-2"><title>What is the diagnosis?</title><sec id="sec2-1"><title></title><sec id="sec3-1"><title>Diagnosis: Vulvo-perineal Crohn's disease</title></sec></sec></sec><sec sec-type="discussion" id="sec1-3"><title>Discussion</title><p>Our patient underwent colonoscopy and small bowel enterography showing no Crohn's disease aspect. Histological examination from intestinal biopsies is also with normal aspect. Based on the clinical (vulvar lymphedema with typical linear inguinal and buttocks fold ulcerations) and histological data (granulomatous histological aspect without caseation), the diagnosis of metastatic Crohn's disease without digestive involvement was obtained. The patient was started with metronidazole 1 g/day. After a 6-month treatment, there was an almost complete healing of ulcerations.</p><p>Vulvo-perineal involvement in Crohn's disease is rare and may constitute the sole manifestation of the disease since it may precede the bowel symptoms by several months to years in 20% of cases and may constitute a diagnostic challenge. Abscesses, draining sinuses, edema, and sharply demarcated ulceration of the perineum and/or vulva with usually verrucous borders (as demonstrated by our case) are the characteristic cutaneous manifestations of the disease. They are caused by direct extension from the involved bowel or by granulomas separated from the bowel by normal tissue. The latter form is usually named metastatic Crohn's disease. In the absence of digestive involvement, the diagnosis is based on clinical and histological aspects and on the absence of other clinical and paraclinical signs of a specific infection.</p><p>The differential diagnosis of metastatic Crohn's disease includes cutaneous tuberculosis, atypical mycobacteriosis, and pemphigus vulgaris especially the vegetated form of pemphigus vulgaris. These diagnoses are eliminated in our case. Indeed, microbiological studies, so as the direct immunofluorescence on cutaneous biopsy, were negative.</p><p>Because of the scarcity of metastatic Crohn's disease and in the absence of a randomized assay, treatment remains not standardized.[<xref ref-type="bibr" rid="ref1">1</xref>] Multiple treatments have been tried. They include corticosteroids which can be administrated either topically or locally injected or orally ingested. They are often efficient with a frequent cortico-dependence and multiple side effects especially for oral steroids. Sulfasalazine may lead to a partial healing of cutaneous lesions, but is more efficient in digestive forms. Immunosuppressive agents such as azathioprine, methotrexate, and cyclosporine are also efficient but a long-term treatment may expose the patient to severe side effects. Immunosuppressive treatments should be indicated in digestive involvement and may be an ultimate option in resistant or severe forms of metastatic Crohn's disease.[<xref ref-type="bibr" rid="ref2">2</xref>] Thalidomide also led to a satisfactory result in refractory vulvar ulcerations associated with Crohn's disease.[<xref ref-type="bibr" rid="ref3">3</xref>] Recently, Prestonet al stated that infliximab is a good therapeutic option for recalcitrant vulvar Crohn's disease but no optimum regimen was provided.[<xref ref-type="bibr" rid="ref4">4</xref>] Metronidazole seems to be a good therapeutic option since it has led in our patient and in several uncontrolled reports to complete healing of perineal lesions, with a persistent result.[<xref ref-type="bibr" rid="ref2">2</xref><xref ref-type="bibr" rid="ref5">5</xref>] Its efficiency is due to antibacterial, anti-inflammatory, and immunosuppressive actions.</p><p>Pelvic manifestations of Crohn's disease can be psychologically crippling. To optimize management, physicians must be aware of its diverse manifestations and confusing presentations.</p></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><ref-list><title>References</title><ref id="ref1"><label>1</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Guest</surname><given-names>GD</given-names></name><name><surname>Fink</surname><given-names>RL</given-names></name></person-group><article-title>Metastatic Crohn's disease: case report of an unusual variant and review of the literature</article-title><source>Dis Colon Rectum</source><year>2000</year><volume>43</volume><fpage>1764</fpage><lpage>6</lpage><pub-id pub-id-type="pmid">11156465</pub-id></element-citation></ref><ref id="ref2"><label>2</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Conscience</surname><given-names>I</given-names></name><name><surname>Perceau</surname><given-names>G</given-names></name><name><surname>Durlach</surname><given-names>A</given-names></name><name><surname>Bernard</surname><given-names>P</given-names></name></person-group><article-title>Vulvar involvement in Crohn's disease: efficacy of metronidazole</article-title><source>Ann Dermatol Venereol</source><year>2006</year><volume>133</volume><fpage>585</fpage><lpage>7</lpage><pub-id pub-id-type="pmid">16885851</pub-id></element-citation></ref><ref id="ref3"><label>3</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Kolivras</surname><given-names>A</given-names></name><name><surname>De Maubeuge</surname><given-names>J</given-names></name><name><surname>André</surname><given-names>J</given-names></name><name><surname>Song</surname><given-names>M</given-names></name></person-group><article-title>Thalidomide in refractory vulvar ulcerations associated with Crohn's disease</article-title><source>Dermatology</source><year>2003</year><volume>206</volume><fpage>381</fpage><lpage>3</lpage><pub-id pub-id-type="pmid">12771492</pub-id></element-citation></ref><ref id="ref4"><label>4</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Preston</surname><given-names>PW</given-names></name><name><surname>Hudson</surname><given-names>N</given-names></name><name><surname>Lewis</surname><given-names>FM</given-names></name></person-group><article-title>Treatment of vulval Crohn's disease with infliximab</article-title><source>Clin Exp Dermatol</source><year>2006</year><volume>31</volume><fpage>378</fpage><lpage>80</lpage><pub-id pub-id-type="pmid">16681581</pub-id></element-citation></ref><ref id="ref5"><label>5</label><element-citation publication-type="journal"><person-group person-group-type="author"><name><surname>Bernstein</surname><given-names>LH</given-names></name><name><surname>Frank</surname><given-names>MS</given-names></name><name><surname>Brandt</surname><given-names>LJ</given-names></name><name><surname>Boley</surname><given-names>SJ</given-names></name></person-group><article-title>Healing of perineal Crohn's disease with metronidazole</article-title><source>Gastroenterology</source><year>1980</year><volume>79</volume><fpage>599</fpage><pub-id pub-id-type="pmid">7429123</pub-id></element-citation></ref></ref-list></back></pmc></record>Pour manipuler ce document sous Unix (Dilib)
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