Rare case of Langerhans cell sarcoma with cutaneous manifestation arising on the inguinal region

Jimura, Nozomi; Matsushita, Satoru; Baba, Naoko; Kubo, Hidemichi; Takeda, Kazuya; Fukushige, Tomoko; Fujii, Kazuyasu; Kanekura, Takuro

doi:10.1111/1346-8138.12692

Cited by 5 publications

(3 citation statements)

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“…A review by Wang et al revealed 37 patients with LCS; in 30 patients with adequate follow‐up 18 (49%) patients died of LCS and 12 (32%) patients had a complete remission following chemotherapy, radiotherapy, surgery, or a combination of these treatments 3 . None of the 18 patients that died of disease presented with skin lesions, while 3 patients who presented only with cutaneous involvement reached complete remission, suggesting that patients with skin lesions may have a relatively better prognosis 4 . In addition, a study by Kawase et al suggested that LCS cases with CD56 expression exhibit a more aggressive behavior 5 …”

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confidence: 99%

“…3 None of the 18 patients that died of disease presented with skin lesions, while 3 patients who presented only with cutaneous involvement reached complete remission, suggesting that patients with skin lesions may have a relatively better prognosis. 4 In addition, a study by Kawase et al suggested that LCS cases with CD56 expression exhibit a more aggressive behavior. 5 Immunohistochemical studies showed that the neoplastic cells were diffusely positive for S100 protein, CD1a, and langerin (CD207) ( Figure 3A-C), and were negative for HMB45, Melan-A, tyrosinase, SOX10, BRAF V600E, pankeratin cocktail, CD117, and CD30.…”

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Langerhans cell sarcoma involving skin and showing epidermotropism: A comprehensive review

et al. 2020

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Langerhans cell sarcoma (LCS) is rare and aggressive; patients have an overall survival rate of less than 50%. We present a 62-year-old man with a history of superficial spreading melanoma of the upper back with sentinel lymph node metastasis, Langerhans cell histiocytosis, and LCS. The patient presented with erythematous papules and scaly areas on his face, neck, arms, chest, abdomen, and legs. A skin biopsy revealed a proliferation of large neoplastic cells involving the dermis and with epidermotropism. These cells had atypical bean-shaped nuclei, with ample cytoplasm and abundant mitotic figures including atypical forms. Immunohistochemical studies showed the tumor to be diffusely positive for CD1a, S100 protein, and langerin (CD207) and negative for melanocytic markers. Some tumor cells were positive for cyclin D1. A diagnosis of LCS involving the skin was established. The present study is a very unusual case of LCS showing epidermotropism. The patient's history of metastatic melanoma posed additional challenges for diagnosis, underlying the need of immunophenotyping in these cases. Consensus for optimal standard therapy has not been established in LCS, and thus, early recognition is important since these neoplasms tend to recur and metastasize. LCS in skin is discussed and published cases are comprehensively reviewed.

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Langerhans cell sarcoma involving skin and showing epidermotropism: A comprehensive review

et al. 2020

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“…It is associated with a high mortality rate even today. LCS, like HS, is extremely rare and is usually diagnosed at an already advanced clinical stage, and it is also associated with a high mortality rate ( 10 ).…”

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confidence: 99%