Granulomatous Lymphangiitis (Melkersson‐Rosenthal Type)
Identifieur interne : 008381 ( Main/Exploration ); précédent : 008380; suivant : 008382Granulomatous Lymphangiitis (Melkersson‐Rosenthal Type)
Auteurs : V. Petronic-Rosic [États-Unis] ; H. Sattar [États-Unis] ; L. White [États-Unis] ; A. Laumann [États-Unis] ; T. Krausz [États-Unis] ; C. R. Shea [États-Unis]Source :
- Journal of Cutaneous Pathology [ 0303-6987 ] ; 2005-01.
Abstract
A 33‐year‐old Caucasian female presented with a ten‐year history of flushing and facial swelling partially responsive to anti‐inflammatory and anti‐microbial therapies. Progressive edema of the right upper eyelid was recently treated by blepharoplasty. The blepharoplasty specimen had a dense mononuclear inflammatory infiltrate and diffuse non‐caseating granulomas composed of epithelioid and multinucleated histiocytes. The granulomas appeared both peri‐and intralymphatic with prominent molding. Special stains and polarization studies were negative. To confirm the intralymphatic location of these granulomas we performed immunohistochemical staining with Factor VIII and D2–40. D2–40 is a new monoclonal antibody that reacts with an O‐linked sialoglycoprotein found on lymphatic endothelium, fetal testis, and on the surface of testicular germ cell tumors. In recent studies, clone D2–40 has shown staining in lymphatic channel endothelium but not in adjacent capillary. Factor VIII antibody highlighted both lymphatic and blood vessel endothelium, while D2–40 stained only the lymphatic endothelium and confirmed the intra‐lymphatic location of granulomas. The patient was diagnosed with granulomatous lymphangiitis of the Melkersson‐Rosenthal type (orofacial granulomatosis). Granulomatous lymphangiitis is characteristic but not completely diagnostic of the Melkersson‐Rosenthal syndrome. We present the complex differential diagnosis of intralymphatic granulomas and the use of a new monoclonal antibody helpful in verifying their location.
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DOI: 10.1111/j.0303-6987.2005.320fm.x
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Shea</name><affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Section of Dermatology and Department of Pathology, The University of Chicago, Chicago, IL</wicri:regionArea><placeName><region type="state">Illinois</region></placeName></affiliation></author></analytic><monogr></monogr><series><title level="j" type="main">Journal of Cutaneous Pathology</title><title level="j" type="alt">JOURNAL OF CUTANEOUS PATHOLOGY</title><idno type="ISSN">0303-6987</idno><idno type="eISSN">1600-0560</idno><imprint><biblScope unit="vol">32</biblScope><biblScope unit="issue">1</biblScope><biblScope unit="page" from="108">108</biblScope><biblScope unit="page" to="109">109</biblScope><biblScope unit="page-count">2</biblScope><publisher>Blackwell Publishing Ltd/Inc.</publisher><pubPlace>Oxford, UK; Malden, USA</pubPlace><date type="published" when="2005-01">2005-01</date></imprint><idno type="ISSN">0303-6987</idno></series></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0303-6987</idno></seriesStmt></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">A 33‐year‐old Caucasian female presented with a ten‐year history of flushing and facial swelling partially responsive to anti‐inflammatory and anti‐microbial therapies. Progressive edema of the right upper eyelid was recently treated by blepharoplasty. The blepharoplasty specimen had a dense mononuclear inflammatory infiltrate and diffuse non‐caseating granulomas composed of epithelioid and multinucleated histiocytes. The granulomas appeared both peri‐and intralymphatic with prominent molding. Special stains and polarization studies were negative. To confirm the intralymphatic location of these granulomas we performed immunohistochemical staining with Factor VIII and D2–40. D2–40 is a new monoclonal antibody that reacts with an O‐linked sialoglycoprotein found on lymphatic endothelium, fetal testis, and on the surface of testicular germ cell tumors. In recent studies, clone D2–40 has shown staining in lymphatic channel endothelium but not in adjacent capillary. Factor VIII antibody highlighted both lymphatic and blood vessel endothelium, while D2–40 stained only the lymphatic endothelium and confirmed the intra‐lymphatic location of granulomas. The patient was diagnosed with granulomatous lymphangiitis of the Melkersson‐Rosenthal type (orofacial granulomatosis). Granulomatous lymphangiitis is characteristic but not completely diagnostic of the Melkersson‐Rosenthal syndrome. We present the complex differential diagnosis of intralymphatic granulomas and the use of a new monoclonal antibody helpful in verifying their location.</div></front></TEI><affiliations><list><country><li>États-Unis</li></country><region><li>Illinois</li></region></list><tree><country name="États-Unis"><region name="Illinois"><name sortKey="Petronic Osic, V" sort="Petronic Osic, V" uniqKey="Petronic Osic V" first="V." last="Petronic-Rosic">V. Petronic-Rosic</name></region><name sortKey="Krausz, T" sort="Krausz, T" uniqKey="Krausz T" first="T." last="Krausz">T. Krausz</name><name sortKey="Laumann, A" sort="Laumann, A" uniqKey="Laumann A" first="A." last="Laumann">A. Laumann</name><name sortKey="Sattar, H" sort="Sattar, H" uniqKey="Sattar H" first="H." last="Sattar">H. Sattar</name><name sortKey="Shea, C R" sort="Shea, C R" uniqKey="Shea C" first="C. R." last="Shea">C. R. Shea</name><name sortKey="White, L" sort="White, L" uniqKey="White L" first="L." last="White">L. White</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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