Persistent lymphoedema in Morbihan disease: formation of perilymphatic epithelioid cell granulomas as a possible pathogenesis
Identifieur interne : 003163 ( Istex/Corpus ); précédent : 003162; suivant : 003164Persistent lymphoedema in Morbihan disease: formation of perilymphatic epithelioid cell granulomas as a possible pathogenesis
Auteurs : T. Nagasaka ; T. Koyama ; K. Matsumura ; K. R. ChenSource :
- Clinical and Experimental Dermatology [ 0307-6938 ] ; 2008-11.
Abstract
Morbihan disease is a rare complication of rosacea, characterized by persistent lymphoedema on the upper half of the face, occurring during the chronic clinical course of rosacea. This refractory condition has been also designated as ‘rosacea lymphoedema’ and ‘solid persistent facial oedema of rosacea’. We report a patient with Morbihan disease showing persistent lymphoedema on the upper half of the face accompanied by unique histological findings of striking dermal dilated lymphatics and damage of the lymphatics at the site of the adjacent epithelioid cell granulomas, with histiocytes bulging into the lymphatic lumen. The marked epithelioid cell granulomas forming around dermal lymphatic vessels with subsequent lymphatic damage and luminal obstruction by histiocytic infiltration may account for the development of lymphoedema in this patient.
Url:
DOI: 10.1111/j.1365-2230.2008.02892.x
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This refractory condition has been also designated as ‘rosacea lymphoedema’ and ‘solid persistent facial oedema of rosacea’. We report a patient with Morbihan disease showing persistent lymphoedema on the upper half of the face accompanied by unique histological findings of striking dermal dilated lymphatics and damage of the lymphatics at the site of the adjacent epithelioid cell granulomas, with histiocytes bulging into the lymphatic lumen. The marked epithelioid cell granulomas forming around dermal lymphatic vessels with subsequent lymphatic damage and luminal obstruction by histiocytic infiltration may account for the development of lymphoedema in this patient.</div></front></TEI><istex><corpusName>wiley</corpusName><keywords><teeft><json:string>rosacea</json:string><json:string>granuloma</json:string><json:string>lymphoedema</json:string><json:string>morbihan</json:string><json:string>dermal</json:string><json:string>lymphatics</json:string><json:string>dermatology</json:string><json:string>morbihan disease</json:string><json:string>persistent lymphoedema</json:string><json:string>oedema</json:string><json:string>histological</json:string><json:string>lymphatic</json:string><json:string>dermatol</json:string><json:string>perilymphatic</json:string><json:string>epithelioid</json:string><json:string>perilymphatic granulomas</json:string><json:string>histiocyte</json:string><json:string>nagasaka</json:string><json:string>ndings</json:string><json:string>histiocytosis</json:string><json:string>dermatologist</json:string><json:string>experimental dermatology</json:string><json:string>dermis</json:string><json:string>obstruction</json:string><json:string>periorbital</json:string><json:string>papule</json:string><json:string>journal compilation</json:string><json:string>british association</json:string><json:string>facial oedema</json:string><json:string>rosacea lymphoedema</json:string><json:string>upper half</json:string><json:string>dermal lymphatics</json:string><json:string>eyelid</json:string></teeft></keywords><author><json:item><name>T. Nagasaka</name><affiliations><json:string>Department of Dermatology, Saiseikai Central Hospital, Tokyo, Japan</json:string></affiliations></json:item><json:item><name>T. Koyama</name><affiliations><json:string>Department of Dermatology, Saiseikai Central Hospital, Tokyo, Japan</json:string></affiliations></json:item><json:item><name>K. Matsumura</name><affiliations><json:string>Department of Dermatology, Saiseikai Central Hospital, Tokyo, Japan</json:string></affiliations></json:item><json:item><name>K. R. 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This refractory condition has been also designated as ‘rosacea lymphoedema’ and ‘solid persistent facial oedema of rosacea’. We report a patient with Morbihan disease showing persistent lymphoedema on the upper half of the face accompanied by unique histological findings of striking dermal dilated lymphatics and damage of the lymphatics at the site of the adjacent epithelioid cell granulomas, with histiocytes bulging into the lymphatic lumen. 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Journal compilation © 2008 British Association of Dermatologists</copyright><eventGroup><event type="firstOnline" date="2008-07-08"></event><event type="publishedOnlineFinalForm" date="2008-10-06"></event><event type="xmlConverted" agent="Converter:BPG_TO_WML3G version:2.3.5 mode:FullText source:FullText result:FullText" date="2010-04-07"></event><event type="xmlConverted" agent="Converter:WILEY_ML3G_TO_WILEY_ML3GV2 version:3.8.8" date="2014-01-09"></event><event type="xmlConverted" agent="Converter:WML3G_To_WML3G version:4.1.7 mode:FullText,remove_FC" date="2014-10-16"></event></eventGroup><numberingGroup><numbering type="pageFirst" number="764">764</numbering><numbering type="pageLast" number="767">767</numbering></numberingGroup><correspondenceTo>Dr Takeshi Nagasaka, Department of Dermatology, Saiseikai Central Hospital, 1‐4‐17 Mita, Minato‐ku, Tokyo 108‐0073, Japan.
E‐mail: <email>takeshingsk@n07.itscom.net</email></correspondenceTo><linkGroup><link type="toTypesetVersion" href="file:CED.CED2892.pdf"></link></linkGroup></publicationMeta><contentMeta><unparsedEditorialHistory>Accepted for publication 7 February 2008</unparsedEditorialHistory><countGroup><count type="figureTotal" number="2"></count><count type="tableTotal" number="0"></count></countGroup><titleGroup><title type="main">Persistent lymphoedema in Morbihan disease: formation of perilymphatic epithelioid cell granulomas as a possible pathogenesis</title><title type="shortAuthors"><i>T. Nagasaka et al.</i></title><title type="short">Persistent lymphoedema in Morbihan disease</title></titleGroup><creators><creator creatorRole="author" xml:id="cr1" affiliationRef="#aff-1-1"><personName><givenNames>T.</givenNames><familyName>Nagasaka</familyName></personName></creator><creator creatorRole="author" xml:id="cr2" affiliationRef="#aff-1-1"><personName><givenNames>T.</givenNames><familyName>Koyama</familyName></personName></creator><creator creatorRole="author" xml:id="cr3" affiliationRef="#aff-1-1"><personName><givenNames>K.</givenNames><familyName>Matsumura</familyName></personName></creator><creator creatorRole="author" xml:id="cr4" affiliationRef="#aff-1-1"><personName><givenNames>K. R.</givenNames><familyName>Chen</familyName></personName></creator></creators><affiliationGroup><affiliation xml:id="aff-1-1" countryCode="JP"><unparsedAffiliation>Department of Dermatology, Saiseikai Central Hospital, Tokyo, Japan</unparsedAffiliation></affiliation></affiliationGroup><abstractGroup><abstract type="main" xml:lang="en"><title type="main">Summary</title><p>Morbihan disease is a rare complication of rosacea, characterized by persistent lymphoedema on the upper half of the face, occurring during the chronic clinical course of rosacea. This refractory condition has been also designated as ‘rosacea lymphoedema’ and ‘solid persistent facial oedema of rosacea’. We report a patient with Morbihan disease showing persistent lymphoedema on the upper half of the face accompanied by unique histological findings of striking dermal dilated lymphatics and damage of the lymphatics at the site of the adjacent epithelioid cell granulomas, with histiocytes bulging into the lymphatic lumen. The marked epithelioid cell granulomas forming around dermal lymphatic vessels with subsequent lymphatic damage and luminal obstruction by histiocytic infiltration may account for the development of lymphoedema in this patient.</p></abstract></abstractGroup></contentMeta><noteGroup><note xml:id="fn1" numbered="no"><p>Conflict of interest: none declared.</p></note></noteGroup></header></component></istex:document></istex:metadataXml><mods version="3.6"><titleInfo lang="en"><title>Persistent lymphoedema in Morbihan disease: formation of perilymphatic epithelioid cell granulomas as a possible pathogenesis</title></titleInfo><titleInfo type="abbreviated" lang="en"><title>Persistent lymphoedema in Morbihan disease</title></titleInfo><titleInfo type="alternative" contentType="CDATA" lang="en"><title>Persistent lymphoedema in Morbihan disease: formation of perilymphatic epithelioid cell granulomas as a possible pathogenesis</title></titleInfo><name type="personal"><namePart type="given">T.</namePart><namePart type="family">Nagasaka</namePart><affiliation>Department of Dermatology, Saiseikai Central Hospital, Tokyo, Japan</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">T.</namePart><namePart type="family">Koyama</namePart><affiliation>Department of Dermatology, Saiseikai Central Hospital, Tokyo, Japan</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">K.</namePart><namePart type="family">Matsumura</namePart><affiliation>Department of Dermatology, Saiseikai Central Hospital, Tokyo, Japan</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">K. R.</namePart><namePart type="family">Chen</namePart><affiliation>Department of Dermatology, Saiseikai Central Hospital, Tokyo, Japan</affiliation><role><roleTerm type="text">author</roleTerm></role></name><typeOfResource>text</typeOfResource><genre type="brief-communication" displayLabel="shortCommunication"></genre><originInfo><publisher>Blackwell Publishing Ltd</publisher><place><placeTerm type="text">Oxford, UK</placeTerm></place><dateIssued encoding="w3cdtf">2008-11</dateIssued><edition>Accepted for publication 7 February 2008</edition><copyrightDate encoding="w3cdtf">2008</copyrightDate></originInfo><language><languageTerm type="code" authority="rfc3066">en</languageTerm><languageTerm type="code" authority="iso639-2b">eng</languageTerm></language><physicalDescription><internetMediaType>text/html</internetMediaType><extent unit="figures">2</extent></physicalDescription><abstract lang="en">Morbihan disease is a rare complication of rosacea, characterized by persistent lymphoedema on the upper half of the face, occurring during the chronic clinical course of rosacea. This refractory condition has been also designated as ‘rosacea lymphoedema’ and ‘solid persistent facial oedema of rosacea’. We report a patient with Morbihan disease showing persistent lymphoedema on the upper half of the face accompanied by unique histological findings of striking dermal dilated lymphatics and damage of the lymphatics at the site of the adjacent epithelioid cell granulomas, with histiocytes bulging into the lymphatic lumen. The marked epithelioid cell granulomas forming around dermal lymphatic vessels with subsequent lymphatic damage and luminal obstruction by histiocytic infiltration may account for the development of lymphoedema in this patient.</abstract><relatedItem type="host"><titleInfo><title>Clinical and Experimental Dermatology</title><subTitle>Experimental dermatology</subTitle></titleInfo><genre type="journal">journal</genre><identifier type="ISSN">0307-6938</identifier><identifier type="eISSN">1365-2230</identifier><identifier type="DOI">10.1111/(ISSN)1365-2230</identifier><identifier type="PublisherID">CED</identifier><part><date>2008</date><detail type="volume"><caption>vol.</caption><number>33</number></detail><detail type="issue"><caption>no.</caption><number>6</number></detail><extent unit="pages"><start>764</start><end>767</end><total>4</total></extent></part></relatedItem><identifier type="istex">6A4091D9E0978EBD2E5EEB3E36FA0EEEED05F006</identifier><identifier type="DOI">10.1111/j.1365-2230.2008.02892.x</identifier><identifier type="ArticleID">CED2892</identifier><accessCondition type="use and reproduction" contentType="copyright">© 2008 The Author(s). 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