Bakry Ola A, Samaka Rehab M, Kandil Mona A, Younes Sheren F
Department of Dermatology, Andrology and STDs.
Rare Tumors. 2013 May 2;5(1):e13. doi: 10.4081/rt.2013.e13. Print 2013 Feb 11.
Histiocytoses are a heterogeneous group of disorders characterized by proliferation and accumulation of cells of mononuclear-macrophage system and dendritic cells. Histiocytoses are categorized according to the cell of origin into Langerhans cell histiocytosis (LCH), Non Langerhans cell histiocytoses and indeterminate cell histiocytosis (ICH). ICH is an extraordinary rare neoplastic dendritic cell disorder that has poorly understood histogenesis and pathogenesis. It is characterized by a proliferation of dendritic cells, which mimic Langerhans cells immunophenotypically (positive for CD1a and S-100 protein), but lack Birbeck granules characteristic of Langerhans cells. Twenty-four year-old Egyptian male was presented with reddish brown chest wall nodule. Clinical, histopathological, immunohistochemical and ultrastructure features are typical for ICH. He was in a good state without any evidence of recurrence or metastasis after 24 months follow up. Peculiar histopathological features were detected in the present case. Many unidentified cells with Hematoxylin & Eosin Langerhans like features showed negative staining for S-100, CD1a, Langerin and CD68. In absence of cellular atypia and mitosis, the infiltrating cells showed epidermotropism that was reported once in ICH as well as neural and perineural invasion that were not previously reported. Therefore we prefer using a tentatively designated diagnosis; dendritic cell tumor, not otherwise specified or newly proposed diagnosis (Indeterminate cell histocytosis with naïve cells) for the present case.
组织细胞增多症是一组异质性疾病,其特征为单核巨噬细胞系统和树突状细胞的增殖和积聚。组织细胞增多症根据起源细胞分为朗格汉斯细胞组织细胞增多症(LCH)、非朗格汉斯细胞组织细胞增多症和不确定细胞组织细胞增多症(ICH)。ICH是一种极其罕见的肿瘤性树突状细胞疾病,其组织发生和发病机制尚不清楚。它的特征是树突状细胞增殖,这些细胞在免疫表型上模仿朗格汉斯细胞(CD1a和S-100蛋白阳性),但缺乏朗格汉斯细胞特有的Birbeck颗粒。一名24岁的埃及男性出现红棕色胸壁结节。临床、组织病理学、免疫组织化学和超微结构特征均为ICH的典型表现。随访24个月后,他状态良好,无复发或转移迹象。本病例检测到特殊的组织病理学特征。许多具有苏木精和伊红染色的类似朗格汉斯细胞特征的未识别细胞,对S-100、CD1a、Langerin和CD68呈阴性染色。在无细胞异型性和有丝分裂的情况下,浸润细胞表现出亲表皮性,这在ICH中曾有过一次报道,以及神经和神经周围侵犯,此前未见报道。因此,对于本病例,我们倾向于使用暂定诊断:未另行指定的树突状细胞瘤或新提出的诊断(含幼稚细胞的不确定细胞组织细胞增多症)。