不确定细胞组织细胞增多症病例报告及文献复习

A. Vojvodić, Z. Mijuskovic, L. Kandolf Sekulovič
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引用次数: 0

摘要

摘要介绍。不确定细胞组织细胞增生症是一种罕见的不确定树突状细胞增生疾病,迄今仅有50例报道。临床表现为黄色、红色或棕色丘疹和结节,出现在其他健康的成年人身上。不确定细胞是皮肤上独特的树突状细胞,其超微结构与表皮朗格汉斯细胞相似,但不含典型的伯贝克颗粒,而且与LCH不同,它们也是朗格汉斯素阴性的。不确定细胞组织细胞病是一种特殊的实体,具有可变的临床,组织病理学或免疫组织化学结果,与朗格汉斯细胞组织细胞病和非朗格汉斯细胞组织细胞病具有相同的形态学和免疫表型特征。病例报告。我们报告一例不确定的组织细胞增多症,患者为77岁男性,3年无症状病史,全身包括口腔黏膜有多个红色和棕色丘疹和结节。行皮肤活检,免疫组化组织病理分析。CD68、CD1a和S-100细胞的脑出血染色阳性使我们能够诊断患者的脑出血。在肿瘤组织中检测到BRAF V600E突变。治疗开始时使用甲氨蝶呤,有效6个月,但由于疾病复发,建议进一步使用沙利度胺治疗,没有效果。结论。不确定性组织细胞增多症是一种罕见的疾病,因此没有建立标准化的治疗方法,治疗选择有限。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
Indeterminate Cell Histiocytosis – Case Report and Review of Literature
Abstract Introduction. Indeterminate cell histiocytosis is a rare proliferative disorder of indeterminate dendritic cells, reported in only 50 case reports so far. It is clinically presented as yellow, red or brown papules and nodules that appear in otherwise healthy adult individuals. Indeterminate cells are distinct dendritic cells of the skin that have ultrastructural similarities to the epidermal Langerhans cells but do not contain the characteristic Birbeck’s granules and they are also langerin-negative, unlike LCH. Indeterminate cell histiocytosis is an exceptional entity with variable clinical, histopathologic or immunohistochemical findings, sharing morphologic and immunophenotypic features with both Langerhans- and non-Langerhans cell histiocytoses. Case Report. We present a case of indeterminate histiocytosis in a 77-year-old man with 3-year history of asymptomatic, multiple reddish and brown papules and nodules over the entire body, including the oral mucosa. Skin biopsy was done, and histopathological analysis with immunohistochemistry was performed. The positive ICH staining of cells for CD68, CD1a, and S-100 enabled us to diagnose ICH in our patient. Also, BRAF V600E mutation was detected in tumor tissue. The treatment was started with methotrexate that was effective for 6 months, but due to the disease recurrence, further therapy with thalidomide was advised, without effect. Conclusion. Indeterminate histiocytosis is a rare disease, therefore no standardized treatment has been established and the treatment options are limited.
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期刊介绍: Serbian Journal of Dermatology and Venereology is a journal of the Serbian Association of Dermatologists and Venereologists. The journal is published in English, quarterly and intended to provide rapid publication of papers in the field of dermatology and venereology. Manuscripts are welcome from all countries in the following categories: editorials, original studies, review articles, professional articles, case reports, and history of medicine.
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