Sarcoidosis of the vulva: A rare cutaneous manifestation
Identifieur interne : 000192 ( Istex/Corpus ); précédent : 000191; suivant : 000193Sarcoidosis of the vulva: A rare cutaneous manifestation
Auteurs : Peter A. Klein ; James Appel ; Jeffrey P. CallenSource :
- Journal of the American Academy of Dermatology [ 0190-9622 ] ; 1998.
English descriptors
- Teeft :
- American academy, Arch dermatol, Brief communications, Callen, Cutaneous, Cutaneous sarcoidosis, Dermatol, Dermatology, Dermatology volume, Granuloma, Lupus pernio, Multinucleated giant cells, Multiple violaceous plaques, Oral chloroquine, Plaque, Right perineum, Sarcoidosis, Slight scale, Solitary plaque, Vulva, Vulval, Vulval involvement, Vulval lesions.
Url:
DOI: 10.1016/S0190-9622(98)70090-8
Links to Exploration step
ISTEX:62C82C4FE14F515AC3D02AF387EC12C622A95B0BLe document en format XML
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Callen, MD, 310 E Broadway, Suite 200, Louisville, KY 40202.</note><note>J Am Acad Dermatol 1998;39:281-3.</note><note>0190-9622/98/$5.00 + 0 16/54/91160</note></notesStmt><sourceDesc><biblStruct type="inbook"><analytic><title level="a" type="main" xml:lang="en">Sarcoidosis of the vulva: A rare cutaneous manifestation</title><author xml:id="author-0000"><persName><forename type="first">Peter A.</forename><surname>Klein</surname></persName><affiliation>Division of Dermatology, Department of Medicine, University of Louisville. Louisville, Kentucky</affiliation></author><author xml:id="author-0001"><persName><forename type="first">James</forename><surname>Appel</surname></persName><roleName type="degree">MD</roleName><affiliation>Division of Dermatology, Department of Medicine, University of Louisville. Louisville, Kentucky</affiliation></author><author xml:id="author-0002"><persName><forename type="first">Jeffrey P.</forename><surname>Callen</surname></persName><roleName type="degree">MD</roleName><affiliation>Division of Dermatology, Department of Medicine, University of Louisville. Louisville, Kentucky</affiliation></author><idno type="istex">62C82C4FE14F515AC3D02AF387EC12C622A95B0B</idno><idno type="ark">ark:/67375/6H6-ZWL86PRZ-W</idno><idno type="DOI">10.1016/S0190-9622(98)70090-8</idno><idno type="PII">S0190-9622(98)70090-8</idno></analytic><monogr><title level="j">Journal of the American Academy of Dermatology</title><title level="j" type="abbrev">YMJD</title><idno type="pISSN">0190-9622</idno><idno type="PII">S0190-9622(05)X7248-6</idno><imprint><publisher>ELSEVIER</publisher><date type="published" when="1998"></date><biblScope unit="volume">39</biblScope><biblScope unit="issue">2</biblScope><biblScope unit="page" from="281">281</biblScope><biblScope unit="page" to="283">283</biblScope></imprint></monogr></biblStruct></sourceDesc></fileDesc><profileDesc><creation><date>1998</date></creation><langUsage><language ident="en">en</language></langUsage></profileDesc><revisionDesc><change when="1998">Published</change></revisionDesc></teiHeader></istex:fulltextTEI><json:item><extension>txt</extension><original>false</original><mimetype>text/plain</mimetype><uri>https://api.istex.fr/ark:/67375/6H6-ZWL86PRZ-W/fulltext.txt</uri></json:item></fulltext><metadata><istex:metadataXml wicri:clean="Elsevier doc found" wicri:toSee="Elsevier, no converted or simple article"><istex:xmlDeclaration>version="1.0" encoding="utf-8"</istex:xmlDeclaration><istex:docType PUBLIC="-//ES//DTD journal article DTD version 5.0.1//EN//XML" URI="art501.dtd" name="istex:docType"><istex:entity SYSTEM="gr1" NDATA="IMAGE" name="gr1"></istex:entity><istex:entity SYSTEM="gr2" NDATA="IMAGE" name="gr2"></istex:entity></istex:docType><istex:document><article docsubtype="fla" xml:lang="en"><item-info><jid>YMJD</jid><aid>91160</aid><ce:pii>S0190-9622(98)70090-8</ce:pii><ce:doi>10.1016/S0190-9622(98)70090-8</ce:doi><ce:copyright type="other" year="1998">American Academy of Dermatology, Inc</ce:copyright></item-info><head><ce:article-footnote><ce:label>☆</ce:label><ce:note-para>Reprint requests: Jeffrey P. Callen, MD, 310 E Broadway, Suite 200, Louisville, KY 40202.</ce:note-para></ce:article-footnote><ce:article-footnote><ce:label>☆☆</ce:label><ce:note-para>J Am Acad Dermatol 1998;39:281-3.</ce:note-para></ce:article-footnote><ce:article-footnote><ce:label>★</ce:label><ce:note-para>0190-9622/98/$5.00 + 0 <ce:bold>16/54/91160</ce:bold></ce:note-para></ce:article-footnote><ce:title>Sarcoidosis of the vulva: A rare cutaneous manifestation</ce:title><ce:author-group><ce:author><ce:given-name>Peter A.</ce:given-name><ce:surname>Klein</ce:surname></ce:author><ce:author><ce:given-name>James</ce:given-name><ce:surname>Appel</ce:surname><ce:degrees>MD</ce:degrees></ce:author><ce:author><ce:given-name>Jeffrey P.</ce:given-name><ce:surname>Callen</ce:surname><ce:degrees>MD</ce:degrees></ce:author><ce:affiliation><ce:textfn>Division of Dermatology, Department of Medicine, University of Louisville. Louisville, Kentucky</ce:textfn></ce:affiliation></ce:author-group></head><body><ce:sections><ce:para id="para0005">Sarcoidosis is a multisystem granulomatous disease of unknown cause affecting the lungs, mediastinal and peripheral lymph nodes, liver, spleen, and skin, with less frequent involvement of the skeletal, cardiac, upper respiratory, and central nervous systems. Intrathoracic involvement has been reported in 83% to 99%<ce:cross-ref refid="bib1"><ce:sup>1</ce:sup></ce:cross-ref> of patients worldwide with a 3% to 6% mortality rate caused by cardiac, pulmonary, or central nervous system disease.<ce:cross-ref refid="bib2"><ce:sup>2</ce:sup></ce:cross-ref> The incidence of sarcoidosis is highest in blacks and Northern European whites with the age at onset typically in the third or fourth decade.</ce:para><ce:para id="para0010">Specific cutaneous manifestations characterized histopathologically by noncaseating granulomas occur in 9% to 37%,<ce:cross-refs refid="bib1 bib3 bib4 bib5 bib6 bib7 bib8 bib9 bib10"><ce:sup>1,3-10</ce:sup></ce:cross-refs> with disease limited to the skin in 27% to 39% of patients.<ce:cross-refs refid="bib11 bib12"><ce:sup>11,12</ce:sup></ce:cross-refs> These manifestations include lupus pernio, infiltrated plaques, maculopapular lesions, subcutaneous nodules, and infiltration of old scars. We describe a case of sarcoidosis of the vulva, which has only been reported twice before.</ce:para><ce:section id="section0005"><ce:section-title>CASE REPORT</ce:section-title><ce:para id="para0015">A 51-year-old black woman had a 3-year history of a persistent pruritic and burning facial and vulval eruption.</ce:para><ce:para id="para0020">Examination revealed a violaceous plaque on the right nasal ala. Multiple violaceous plaques with slight scale were present on the vulva with bilateral perivulval hypopigmented linear plaques (Fig. 1).<ce:display><ce:figure id="figure0005"><ce:label>Fig. 1</ce:label><ce:caption><ce:simple-para id="simple-para0005">Multiple violaceous plaques with slight scale on vulva; solitary plaque on right perineum.</ce:simple-para></ce:caption><ce:link locator="gr1"></ce:link></ce:figure></ce:display>A solitary plaque was also present on the right perineum.</ce:para><ce:para id="para0025">Laboratory evaluation revealed normal blood count and electrolyte profiles. Serum calcium and liver function tests were also within normal limits. The serum angiotensin-converting enzyme level was normal at 31 nmol/min<ce:sup>-1</ce:sup> /ml<ce:sup>-1</ce:sup> . Pulmonary function studies and a chest radiograph were normal. Ophthalmologic examination was normal.</ce:para><ce:para id="para0030">Biopsy specimens of the nasal and vulval skin lesions revealed a nodular noncaseating granulomatous dermatitis with multinucleated giant cells. Some granulomas displayed a thin rim of lymphocytes (Fig. 2).<ce:display><ce:figure id="figure0015"><ce:label>Fig. 2</ce:label><ce:caption><ce:simple-para id="simple-para0015">Nodular noncaseating granulomas with multinucleated giant cells. Some granulomas display thin rim of lymphocytes. (Hematoxylin-eosin stain; original magnification ×40.)</ce:simple-para></ce:caption><ce:link locator="gr2"></ce:link></ce:figure></ce:display>Periodic acid–Schiff and Fite stains were negative for fungi and acid-fast mycobacteria, respectively.</ce:para><ce:para id="para0035">The patient was treated with oral hydroxychloroquine 200 mg twice daily. Because of minimal improvement, she was changed to oral chloroquine 500 mg daily and a mid-potency topical corticosteroid. Four months after initiating therapy, the lesions improved slightly. The patient subsequently received an intralesional injection of triamcinolone acetonide at the right nasal ala, which resulted in flattening of the plaque. The vulval lesions, however, persisted despite continued oral chloroquine.</ce:para></ce:section><ce:section id="section0015"><ce:section-title>DISCUSSION</ce:section-title><ce:para id="para0040">Our patient demonstrates a rare presentation of sarcoidosis limited to the vulval and facial skin in the absence of demonstrable systemic manifestations. It is similar to the first case report in which the patient had cutaneous sarcoidosis of the face, neck, and vulva.<ce:cross-ref refid="bib13"><ce:sup>13</ce:sup></ce:cross-ref> The second report described papular vulval involvement, but bilateral hilar lymphadenopathy was also present; vulval involvement was the presenting manifestation of disease.<ce:cross-ref refid="bib14"><ce:sup>14</ce:sup></ce:cross-ref></ce:para><ce:para id="para0045">Vulval involvement is likely a cutaneous manifestation of clinical sarcoidosis. Unlike erythema nodosum in the setting of Löfgren’s syndrome (anterior uveitis, fever, polyarthralgia, and asymptomatic bilateral hilar adenopathy), which is a characteristic of acute benign disease, cutaneous plaques are an indicator of a chronic process. In patients presenting with plaques, 93% will continue to be affected by sarcoidosis 2 years after diagnosis.<ce:cross-ref refid="bib15"><ce:sup>15</ce:sup></ce:cross-ref></ce:para><ce:para id="para0050">Sarcoidosis has also been reported in the female reproductive tract including the fallopian tubes, ovaries, and uterus.<ce:cross-ref refid="bib16"><ce:sup>16</ce:sup></ce:cross-ref></ce:para><ce:para id="para0055">Therapy for cutaneous sarcoidosis is frequently difficult.<ce:cross-refs refid="bib15 bib17"><ce:sup>15,17</ce:sup></ce:cross-refs> High-dose systemic corticosteroids are effective but result in toxicity. Intralesional triamcinolone injections are frequently helpful and were effective for the facial lesions in our patient. The patient did not wish to undergo this therapy for the vulval lesions. Other options include antimalarials, allopurinol, methotrexate, and chlorambucil.</ce:para></ce:section></ce:sections></body><tail><ce:bibliography id="bibliography0005"><ce:section-title>References</ce:section-title><ce:bibliography-sec id="bibliography-sec0005"><ce:bib-reference id="bib1"><ce:label>1</ce:label><sb:reference><sb:contribution><sb:authors><sb:author><ce:given-name>DG</ce:given-name><ce:surname>James</ce:surname></sb:author><sb:author><ce:given-name>E</ce:given-name><ce:surname>Neville</ce:surname></sb:author><sb:author><ce:given-name>LE.</ce:given-name><ce:surname>Siltzbach</ce:surname></sb:author></sb:authors><sb:title><sb:maintitle>A worldwide review of sarcoidosis</sb:maintitle></sb:title></sb:contribution><sb:host><sb:issue><sb:series><sb:title><sb:maintitle>Ann N Y Acad 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Med</sb:maintitle></sb:title><sb:volume-nr>336</sb:volume-nr></sb:series><sb:date>1997</sb:date></sb:issue><sb:pages><sb:first-page>1224</sb:first-page><sb:last-page>1234</sb:last-page></sb:pages></sb:host></sb:reference></ce:bib-reference></ce:bibliography-sec></ce:bibliography></tail></article></istex:document></istex:metadataXml><mods version="3.6"><titleInfo lang="en"><title>Sarcoidosis of the vulva: A rare cutaneous manifestation</title></titleInfo><titleInfo type="alternative" lang="en" contentType="CDATA"><title>Sarcoidosis of the vulva: A rare cutaneous manifestation</title></titleInfo><name type="personal"><namePart type="given">Peter A.</namePart><namePart type="family">Klein</namePart><affiliation>Division of Dermatology, Department of Medicine, University of Louisville. Louisville, Kentucky</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">James</namePart><namePart type="family">Appel</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Division of Dermatology, Department of Medicine, University of Louisville. Louisville, Kentucky</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Jeffrey P.</namePart><namePart type="family">Callen</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Division of Dermatology, Department of Medicine, University of Louisville. Louisville, Kentucky</affiliation><role><roleTerm type="text">author</roleTerm></role></name><typeOfResource>text</typeOfResource><genre type="research-article" displayLabel="Full-length article" authority="ISTEX" authorityURI="https://content-type.data.istex.fr" valueURI="https://content-type.data.istex.fr/ark:/67375/XTP-1JC4F85T-7">research-article</genre><originInfo><publisher>ELSEVIER</publisher><dateIssued encoding="w3cdtf">1998</dateIssued><copyrightDate encoding="w3cdtf">1998</copyrightDate></originInfo><language><languageTerm type="code" authority="iso639-2b">eng</languageTerm><languageTerm type="code" authority="rfc3066">en</languageTerm></language><note>Reprint requests: Jeffrey P. 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