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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Metastatic Crohn’s disease accompanying granulomatous vasculitis and lymphangitis in the vulva</title>
<author><name sortKey="Ishida, Mitsuaki" sort="Ishida, Mitsuaki" uniqKey="Ishida M" first="Mitsuaki" last="Ishida">Mitsuaki Ishida</name>
</author>
<author><name sortKey="Iwai, Muneo" sort="Iwai, Muneo" uniqKey="Iwai M" first="Muneo" last="Iwai">Muneo Iwai</name>
</author>
<author><name sortKey="Yoshida, Keiko" sort="Yoshida, Keiko" uniqKey="Yoshida K" first="Keiko" last="Yoshida">Keiko Yoshida</name>
</author>
<author><name sortKey="Kagotani, Akiko" sort="Kagotani, Akiko" uniqKey="Kagotani A" first="Akiko" last="Kagotani">Akiko Kagotani</name>
</author>
<author><name sortKey="Okabe, Hidetoshi" sort="Okabe, Hidetoshi" uniqKey="Okabe H" first="Hidetoshi" last="Okabe">Hidetoshi Okabe</name>
</author>
</titleStmt>
<publicationStmt><idno type="wicri:source">PMC</idno>
<idno type="pmid">24133609</idno>
<idno type="pmc">3796253</idno>
<idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3796253</idno>
<idno type="RBID">PMC:3796253</idno>
<date when="2013">2013</date>
<idno type="wicri:Area/Pmc/Corpus">002035</idno>
<idno type="wicri:explorRef" wicri:stream="Pmc" wicri:step="Corpus" wicri:corpus="PMC">002035</idno>
</publicationStmt>
<sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Metastatic Crohn’s disease accompanying granulomatous vasculitis and lymphangitis in the vulva</title>
<author><name sortKey="Ishida, Mitsuaki" sort="Ishida, Mitsuaki" uniqKey="Ishida M" first="Mitsuaki" last="Ishida">Mitsuaki Ishida</name>
</author>
<author><name sortKey="Iwai, Muneo" sort="Iwai, Muneo" uniqKey="Iwai M" first="Muneo" last="Iwai">Muneo Iwai</name>
</author>
<author><name sortKey="Yoshida, Keiko" sort="Yoshida, Keiko" uniqKey="Yoshida K" first="Keiko" last="Yoshida">Keiko Yoshida</name>
</author>
<author><name sortKey="Kagotani, Akiko" sort="Kagotani, Akiko" uniqKey="Kagotani A" first="Akiko" last="Kagotani">Akiko Kagotani</name>
</author>
<author><name sortKey="Okabe, Hidetoshi" sort="Okabe, Hidetoshi" uniqKey="Okabe H" first="Hidetoshi" last="Okabe">Hidetoshi Okabe</name>
</author>
</analytic>
<series><title level="j">International Journal of Clinical and Experimental Pathology</title>
<idno type="eISSN">1936-2625</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>Metastatic Crohn’s disease (CD) is an extremely rare extragastrointestinal manifestation of CD, and is characterized histopathologically by the presence of non-caseating granulomatous inflammation. Granulomatous vasculitis and lymphangitis have rarely been documented in metastatic CD. Herein, we report the first documented case of metastatic CD accompanied by both granulomatous vasculitis and lymphangitis in the vulva. A 35-year-old Japanese female with CD presented with multiple small nodules in her vulva. Biopsy was performed under a clinical diagnosis of genital warts. A histopathological study revealed marked lymphangiectasia in the papillary dermis. Within the dilated lymphatics, lymphocytes and aggregates of macrophages were present, which are typical features of granulomatous lymphangitis. Tiny non-caseating granulomas and granulomatous vasculitis were also observed. Accordingly, a diagnosis of metastatic CD accompanied by both granulomatous vasculitis and lymphangitis was made. The occurrence of cutaneous lesions in patients with CD is well known. Albeit extremely rare, lymphangiectasia has been reported in the vulva of CD patients that clinically mimicked viral warts, as in the present case. The diagnosis of metastatic CD in the present case was not difficult because characteristic histopathological features were present, and a clinical history of CD was available. However, a few cases of genital swelling associated with granulomatous inflammation prior to a diagnosis of gastrointestinal CD have been documented. Therefore, granulomatous vasculitis and lymphangitis in the external genitals should be considered as potential indication of metastatic CD even in cases without a history of gastrointestinal CD.</p>
</div>
</front>
</TEI>
<pmc article-type="case-report"><pmc-comment>The publisher of this article does not allow downloading of the full text in XML form.</pmc-comment>
<front><journal-meta><journal-id journal-id-type="nlm-ta">Int J Clin Exp Pathol</journal-id>
<journal-id journal-id-type="iso-abbrev">Int J Clin Exp Pathol</journal-id>
<journal-id journal-id-type="publisher-id">ijcep</journal-id>
<journal-title-group><journal-title>International Journal of Clinical and Experimental Pathology</journal-title>
</journal-title-group>
<issn pub-type="epub">1936-2625</issn>
<publisher><publisher-name>e-Century Publishing Corporation</publisher-name>
</publisher>
</journal-meta>
<article-meta><article-id pub-id-type="pmid">24133609</article-id>
<article-id pub-id-type="pmc">3796253</article-id>
<article-categories><subj-group subj-group-type="heading"><subject>Case Report</subject>
</subj-group>
</article-categories>
<title-group><article-title>Metastatic Crohn’s disease accompanying granulomatous vasculitis and lymphangitis in the vulva</article-title>
</title-group>
<contrib-group><contrib contrib-type="author"><name><surname>Ishida</surname>
<given-names>Mitsuaki</given-names>
</name>
</contrib>
<contrib contrib-type="author"><name><surname>Iwai</surname>
<given-names>Muneo</given-names>
</name>
</contrib>
<contrib contrib-type="author"><name><surname>Yoshida</surname>
<given-names>Keiko</given-names>
</name>
</contrib>
<contrib contrib-type="author"><name><surname>Kagotani</surname>
<given-names>Akiko</given-names>
</name>
</contrib>
<contrib contrib-type="author"><name><surname>Okabe</surname>
<given-names>Hidetoshi</given-names>
</name>
</contrib>
<aff><institution>Department of Clinical Laboratory Medicine and Division of Diagnostic Pathology, Shiga University of Medical Science</institution>
<addr-line>Shiga, Japan</addr-line>
</aff>
</contrib-group>
<author-notes><corresp><bold>Address correspondence to:</bold>
Dr. Mitsuaki Ishida, Department of Clinical Laboratory Medicine and Division of Diagnostic Pathology, Shiga University of Medical Science, Tsukinowa-cho, Seta, Otsu, Shiga, 520-2192, Japan. Tel: +81-77-548-2603; Fax: +81-77-548-2407; E-mail: <email>mitsuaki@belle.shiga-med.ac.jp</email>
</corresp>
</author-notes>
<pub-date pub-type="collection"><year>2013</year>
</pub-date>
<pub-date pub-type="epub"><day>15</day>
<month>9</month>
<year>2013</year>
</pub-date>
<volume>6</volume>
<issue>10</issue>
<fpage>2263</fpage>
<lpage>2266</lpage>
<history><date date-type="received"><day>22</day>
<month>7</month>
<year>2013</year>
</date>
<date date-type="accepted"><day>15</day>
<month>8</month>
<year>2013</year>
</date>
</history>
<permissions><copyright-statement>IJCEP Copyright © 2013</copyright-statement>
<copyright-year>2013</copyright-year>
</permissions>
<abstract><p>Metastatic Crohn’s disease (CD) is an extremely rare extragastrointestinal manifestation of CD, and is characterized histopathologically by the presence of non-caseating granulomatous inflammation. Granulomatous vasculitis and lymphangitis have rarely been documented in metastatic CD. Herein, we report the first documented case of metastatic CD accompanied by both granulomatous vasculitis and lymphangitis in the vulva. A 35-year-old Japanese female with CD presented with multiple small nodules in her vulva. Biopsy was performed under a clinical diagnosis of genital warts. A histopathological study revealed marked lymphangiectasia in the papillary dermis. Within the dilated lymphatics, lymphocytes and aggregates of macrophages were present, which are typical features of granulomatous lymphangitis. Tiny non-caseating granulomas and granulomatous vasculitis were also observed. Accordingly, a diagnosis of metastatic CD accompanied by both granulomatous vasculitis and lymphangitis was made. The occurrence of cutaneous lesions in patients with CD is well known. Albeit extremely rare, lymphangiectasia has been reported in the vulva of CD patients that clinically mimicked viral warts, as in the present case. The diagnosis of metastatic CD in the present case was not difficult because characteristic histopathological features were present, and a clinical history of CD was available. However, a few cases of genital swelling associated with granulomatous inflammation prior to a diagnosis of gastrointestinal CD have been documented. Therefore, granulomatous vasculitis and lymphangitis in the external genitals should be considered as potential indication of metastatic CD even in cases without a history of gastrointestinal CD.</p>
</abstract>
<kwd-group><kwd>Metastatic Crohn’s disease</kwd>
<kwd>granulomatous vasculitis</kwd>
<kwd>granulomatous lymphangitis</kwd>
<kwd>vulva</kwd>
</kwd-group>
</article-meta>
</front>
</pmc>
</record>
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