Nodular 'pure' mucocutaneous histiocytosis X in an adult

Caputo, R.

doi:10.1001/archderm.123.10.1274

Cited by 7 publications

(6 citation statements)

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“…In adults, it is especially uncommon when the presentation is limited to the skin, as shown in table 1 [4, 5, 6, 7, 8, 9, 10, 11, 12, 13, 14, 15, 16, 17, 18, 19, 20, 21, 22, 23, 24, 25]. …”

Section: Discussionmentioning

confidence: 99%

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Adult Langerhans Cell Histiocytosis Limited to the Skin

Singh

Prieto²,

Czelusta

et al. 2003

Dermatology

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Background: The incidence of Langerhans cell histiocytosis (LCH) is 4–5 per million in children with only 30% of this number having an adult onset. While dermatological manifestations occur in as many as 50% of cases, disease limited to the skin is uncommon among reported cases of adult LCH. Objectives and Methods: To present 3 new cases of adult LCH and a review of the literature of isolated cutaneous LCH in adults. Results: Three adults with scalp, vulvar and generalized LCH lesions had refractory responses to treatment. Conclusions: LCH may present with unusual cutaneous manifestations limited to the skin in adults. Optimal treatment has not yet been determined.

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“…In adults, it is especially uncommon when the presentation is limited to the skin, as shown in table 1 [4, 5, 6, 7, 8, 9, 10, 11, 12, 13, 14, 15, 16, 17, 18, 19, 20, 21, 22, 23, 24, 25]. …”

Section: Discussionmentioning

confidence: 99%

“…The clinical presentation of cutaneous LCH is highly variable [26]and can be generalized papules [10]or nodules [5], localized scalp lesions or ulcers in skin folds [17]. Predominant sites of cutaneous involvement include the scalp, flexural areas, external genitalia and glabrous skin [2, 27, 28].…”

Section: Discussionmentioning

confidence: 99%

Adult Langerhans Cell Histiocytosis Limited to the Skin

Singh

Prieto²,

Czelusta

et al. 2003

Dermatology

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“…Distinguishing between reactive versus intrinsic neoplastic mechanisms is important as the first scenario is self- limited in contrast to the latter which has a relatively poor prognosis and requires systemic chemotherapy. Although there are no reported cases of LCH associated with molluscum contagiosum to date, we found three case reports of patients with molluscum-like papules ultimately diagnosed as having LCH 11–13 . We report our findings in a patient with concurrent solitary cutaneous hyperplasia of Langerhans cells and molluscum confirmed clinically, histologically and by polymerase chain reaction (PCR).…”

Section: Discussionmentioning

confidence: 84%

Langerhans Cell Hyperplasia From Molluscum Contagiosum

Hatter

Zhou

Honda

et al. 2015

The American Journal of Dermatopathology

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Langerhans cell histiocytosis (LCH) carries a prognosis which ranges from benign to potentially fatal. There is currently little framework to decipher metrics which predict the benign versus aggressive nature of LCH. We wanted to determine if molluscum contagiosum virus (MCV) DNA could be isolated from a cutaneous lesion demonstrating Langerhans cell hyperplasia resembling LCH in a patient with both. We performed polymerase chain reaction (PCR) on biopsy proven MCV and the hyperplastic lesion. Two specific regions within the MCV genome were detected from both biopsies. We report our findings and suggest that some MCV can produce histologic lesions resembling LCH, similar to the literature on scabies mimicking LCH. Efforts to find a reactive “driver” in LCH may significantly inform the clinical scenario.

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“…However, patients presenting with generalized papular lesions and/or ulcerating lesions in the skin folds appear much more resistant to treatment. Many agents including topical and systemic corticosteroids, topical nitrogen mustard, imiquimod, narrowband UVB, PUVA, radiotherapy, thalidomide, methotrexate, retinoids, vinblastine, interferon and cladribine have been tried and have been reported successful in some patients 4–67 . However, no single treatment was effective in all cases.…”

Section: Discussionmentioning

confidence: 99%

Langerhans cell histiocytosis first presenting in the skin in adults: frequent association with a second haematological malignancy

Edelbroek¹,

Vermeer²,

Jansen

et al. 2012

British Journal of Dermatology

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Summary Background Langerhans cell histiocytosis (LCH) in adults first presenting in the skin is rare. Guidelines for staging, treatment and follow‐up are lacking. Objectives To better define staging procedures, treatment results and clinical course in adult patients with LCH first presenting in the skin. Methods Eighteen adult patients with LCH first presenting in the skin were collected from five centres collaborating in the Dutch Cutaneous Lymphoma Group. Clinical records and (skin) biopsy specimens were reviewed and follow‐up data were obtained. A literature search on adult patients with LCH presenting in the skin was performed. Results Staging procedures showed extracutaneous disease in three of 16 patients who were adequately staged. One patient had a histologically confirmed lytic LCH bone lesion, while two patients had a myelodysplastic syndrome. During follow‐up two of 18 patients developed extracutaneous localizations of LCH. Five patients developed a second haematological malignancy, including (myelo)monocytic leukaemia (two cases), histiocytic sarcoma (one case), diffuse large B‐cell lymphoma (one case) and peripheral T‐cell lymphoma (one case). Review of the literature revealed six other adult patients with a second haematological malignancy preceding or following a diagnosis of LCH. Conclusions The results of the present study suggest an increased risk of a second haematological malignancy in adult patients with LCH presenting in the skin. Extensive staging at presentation and long‐term follow‐up are therefore warranted in such patients.

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Nodular 'pure' mucocutaneous histiocytosis X in an adult

Cited by 7 publications

References 0 publications

Adult Langerhans Cell Histiocytosis Limited to the Skin

Adult Langerhans Cell Histiocytosis Limited to the Skin

Langerhans Cell Hyperplasia From Molluscum Contagiosum

Langerhans cell histiocytosis first presenting in the skin in adults: frequent association with a second haematological malignancy

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