Persistent lymphoedema in Morbihan disease: formation of perilymphatic epithelioid cell granulomas as a possible pathogenesis.
Identifieur interne : 003200 ( PubMed/Corpus ); précédent : 003199; suivant : 003201Persistent 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 [ 1365-2230 ] ; 2008.
English descriptors
- KwdEn :
- MESH :
- immunology : Epithelioid Cells, Facial Dermatoses, Granuloma, Histiocytes, Lymphatic Vessels, Lymphedema, Rosacea, Skin.
- Aged, Humans, Male, Syndrome.
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.
DOI: 10.1111/j.1365-2230.2008.02892.x
PubMed: 18627384
Links to Exploration step
pubmed:18627384Le document en format XML
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<author><name sortKey="Nagasaka, T" sort="Nagasaka, T" uniqKey="Nagasaka T" first="T" last="Nagasaka">T. Nagasaka</name>
<affiliation><nlm:affiliation>Department of Dermatology, Saiseikai Central Hospital, Tokyo, Japan. takeshingsk@n07.itscom.net</nlm:affiliation>
</affiliation>
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<author><name sortKey="Koyama, T" sort="Koyama, T" uniqKey="Koyama T" first="T" last="Koyama">T. Koyama</name>
</author>
<author><name sortKey="Matsumura, K" sort="Matsumura, K" uniqKey="Matsumura K" first="K" last="Matsumura">K. Matsumura</name>
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<author><name sortKey="Chen, K R" sort="Chen, K R" uniqKey="Chen K" first="K R" last="Chen">K R Chen</name>
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<sourceDesc><biblStruct><analytic><title xml:lang="en">Persistent lymphoedema in Morbihan disease: formation of perilymphatic epithelioid cell granulomas as a possible pathogenesis.</title>
<author><name sortKey="Nagasaka, T" sort="Nagasaka, T" uniqKey="Nagasaka T" first="T" last="Nagasaka">T. Nagasaka</name>
<affiliation><nlm:affiliation>Department of Dermatology, Saiseikai Central Hospital, Tokyo, Japan. takeshingsk@n07.itscom.net</nlm:affiliation>
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<author><name sortKey="Koyama, T" sort="Koyama, T" uniqKey="Koyama T" first="T" last="Koyama">T. Koyama</name>
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<author><name sortKey="Matsumura, K" sort="Matsumura, K" uniqKey="Matsumura K" first="K" last="Matsumura">K. Matsumura</name>
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<author><name sortKey="Chen, K R" sort="Chen, K R" uniqKey="Chen K" first="K R" last="Chen">K R Chen</name>
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<series><title level="j">Clinical and experimental dermatology</title>
<idno type="eISSN">1365-2230</idno>
<imprint><date when="2008" type="published">2008</date>
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<profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Aged</term>
<term>Epithelioid Cells (immunology)</term>
<term>Facial Dermatoses (immunology)</term>
<term>Granuloma (immunology)</term>
<term>Histiocytes (immunology)</term>
<term>Humans</term>
<term>Lymphatic Vessels (immunology)</term>
<term>Lymphedema (immunology)</term>
<term>Male</term>
<term>Rosacea (immunology)</term>
<term>Skin (immunology)</term>
<term>Syndrome</term>
</keywords>
<keywords scheme="MESH" qualifier="immunology" xml:lang="en"><term>Epithelioid Cells</term>
<term>Facial Dermatoses</term>
<term>Granuloma</term>
<term>Histiocytes</term>
<term>Lymphatic Vessels</term>
<term>Lymphedema</term>
<term>Rosacea</term>
<term>Skin</term>
</keywords>
<keywords scheme="MESH" xml:lang="en"><term>Aged</term>
<term>Humans</term>
<term>Male</term>
<term>Syndrome</term>
</keywords>
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<front><div type="abstract" xml: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.</div>
</front>
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<DateCreated><Year>2008</Year>
<Month>10</Month>
<Day>28</Day>
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<DateCompleted><Year>2008</Year>
<Month>12</Month>
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<DateRevised><Year>2008</Year>
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<Article PubModel="Print-Electronic"><Journal><ISSN IssnType="Electronic">1365-2230</ISSN>
<JournalIssue CitedMedium="Internet"><Volume>33</Volume>
<Issue>6</Issue>
<PubDate><Year>2008</Year>
<Month>Nov</Month>
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<Title>Clinical and experimental dermatology</Title>
<ISOAbbreviation>Clin. Exp. Dermatol.</ISOAbbreviation>
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<ArticleTitle>Persistent lymphoedema in Morbihan disease: formation of perilymphatic epithelioid cell granulomas as a possible pathogenesis.</ArticleTitle>
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<Abstract><AbstractText>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.</AbstractText>
</Abstract>
<AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Nagasaka</LastName>
<ForeName>T</ForeName>
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<AffiliationInfo><Affiliation>Department of Dermatology, Saiseikai Central Hospital, Tokyo, Japan. takeshingsk@n07.itscom.net</Affiliation>
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<Author ValidYN="Y"><LastName>Koyama</LastName>
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<Language>eng</Language>
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<MeshHeadingList><MeshHeading><DescriptorName UI="D000368" MajorTopicYN="N">Aged</DescriptorName>
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<MeshHeading><DescriptorName UI="D015622" MajorTopicYN="N">Epithelioid Cells</DescriptorName>
<QualifierName UI="Q000276" MajorTopicYN="Y">immunology</QualifierName>
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<MeshHeading><DescriptorName UI="D005148" MajorTopicYN="N">Facial Dermatoses</DescriptorName>
<QualifierName UI="Q000276" MajorTopicYN="Y">immunology</QualifierName>
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<MeshHeading><DescriptorName UI="D006099" MajorTopicYN="N">Granuloma</DescriptorName>
<QualifierName UI="Q000276" MajorTopicYN="Y">immunology</QualifierName>
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<MeshHeading><DescriptorName UI="D006644" MajorTopicYN="N">Histiocytes</DescriptorName>
<QualifierName UI="Q000276" MajorTopicYN="N">immunology</QualifierName>
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<MeshHeading><DescriptorName UI="D006801" MajorTopicYN="N">Humans</DescriptorName>
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<MeshHeading><DescriptorName UI="D042601" MajorTopicYN="N">Lymphatic Vessels</DescriptorName>
<QualifierName UI="Q000276" MajorTopicYN="N">immunology</QualifierName>
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<MeshHeading><DescriptorName UI="D008209" MajorTopicYN="N">Lymphedema</DescriptorName>
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<MeshHeading><DescriptorName UI="D008297" MajorTopicYN="N">Male</DescriptorName>
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<MeshHeading><DescriptorName UI="D012393" MajorTopicYN="N">Rosacea</DescriptorName>
<QualifierName UI="Q000276" MajorTopicYN="Y">immunology</QualifierName>
</MeshHeading>
<MeshHeading><DescriptorName UI="D012867" MajorTopicYN="N">Skin</DescriptorName>
<QualifierName UI="Q000276" MajorTopicYN="N">immunology</QualifierName>
</MeshHeading>
<MeshHeading><DescriptorName UI="D013577" MajorTopicYN="N">Syndrome</DescriptorName>
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