The "Eosinophilic Granulomas" of the Skin

Weidman, Fred D.

doi:10.1001/archderm.1947.01520020002001

Cited by 60 publications

(12 citation statements)

References 4 publications

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“…In our review of the literature we identified only 16 cases of LCH initially presenting with isolated vulvar disease (Tables 1 and 2), with the first case of isolated vulvar LCH reported in 1947 (4). We also identified other case reports of socalled isolated vulvar LCH in the literature.…”

Section: Discussionmentioning

confidence: 99%

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Isolated Vulvar Langerhans Cell Histiocytosis: Report of Two Cases

Padula

Medeiros²,

Silva

et al. 2004

International Journal of Gynecological Pathology

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Two cases of Langerhans cell histiocytosis involving the vulva are reported. The clinical features of Case 1 have been previously reported. The patients, aged 31 (Case 1) and 52 (Case 2) years, had disease limited to the vulva at the time of diagnosis. In both cases, the vulvar lesions were composed of Langerhans cells with twisted nuclei and nuclear grooves, associated with eosinophils and other inflammatory cells. Immunohistochemical studies using fixed, paraffin-embedded tissue sections showed that the Langerhans cells were positive for CD1A and S-100 protein in both cases, supporting the morphologic diagnosis. The first patient had multiple local recurrences during 3 years despite radiation therapy and vulvectomy, but is currently in remission and being treated with thalidomide. The second patient experienced disseminated disease involving multiple bony sites and the mouth despite radiation therapy. Including the present two cases, 6 of 18 (33%) cases of isolated vulvar LCH subsequently disseminated, most commonly to bone; other patients had local recurrences. Thus isolated vulvar LCH has the potential for aggressive clinical behavior, either as local recurrence or disseminated disease. New treatment modalities are needed for this disease.

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Section: Discussionmentioning

confidence: 99%

“…To date, 16 cases of isolated (localized) vulvar LCH have been reported (4,(6)(7)(8)(9)(10)(11)(12)(13)(14)(15)(16)(17). In five of these cases, LCH subsequently disseminated to other sites (7,(15)(16)(17).…”

mentioning

confidence: 99%

Isolated Vulvar Langerhans Cell Histiocytosis: Report of Two Cases

Padula

Medeiros²,

Silva

et al. 2004

International Journal of Gynecological Pathology

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“…8 There have been only 19 case reports of primary isolated vulval adult LCH with the youngest affected patient being 16.5 years old as summarized in Table 3. [1][2][3][6][7][8][9]11,15,16,[19][20][21][22][23] Vulval ulcers are the most common presentation of genital LCH. 10 Genital LCH can also present as erythematous plaques, papules or nodules.…”

Section: Discussionmentioning

confidence: 99%

Rapid resolution of primary vulval adult Langerhans cell histiocytosis with very potent topical corticosteroids

Foley¹,

Panting²,

Bell³

et al. 2011

Australasian Journal of Dermatology

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Langerhans cell histiocytosis is a rare idiopathic disorder with protean clinical presentations. Primary unifocal single-system disease of the vulva is even less common. We report a 62-year-old female patient presenting with an 18-month history of pruritus and burning of the vulva. Clinical examination of the vulva showed a tender nodule of the right labium minus. Histology confirmed Langerhans cell histiocytosis. Systemic involvement was excluded. Within 1 month the use of clobetasol propionate ointment led to resolution of both the patient's symptoms and the clinical appearance of the affected right labium minus. This resolution was maintained 12 months later.

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“…In 1947 Weidman [9] reported a patient with Hodgkin's disease of the skin with massive peripheral blood and tissue eosinophilia, the skin and lym ph nodes being particularly involved. It is well known th at eosinophilia is often a feature of lymphomas [10], although it is rarely as massive as it was in this patient, nor does it usually involve the skin.…”

Section: Discussionmentioning

confidence: 99%