Mohsin S K, Lee M W, Amin M B, Stoler M H, Eyzaguirre E, Ma C K, Zarbo R J
Department of Pathology, Henry Ford Hospital, Detroit, Michigan 48202, USA.
Am J Surg Pathol. 1998 Apr;22(4):479-87. doi: 10.1097/00000478-199804000-00014.
Verruciform xanthoma is a rare clinicopathologic entity of uncertain etiology that occurs primarily in the oral mucosa. Aggregates of foam cells in the submucosal stroma or papillary dermis in association with verrucous epithelial hyperplasia are the hallmark of this lesion. Extraoral (cutaneous) occurrence of verruciform xanthoma is much rarer and has been reported mostly in the genital skin. Five cases of extraoral cutaneous verruciform xanthoma (three from the scrotum, one from the penis, and one from the nose) and one histologic "simulant" (from skin of the nose) were studied. The lesions were solitary, raised, or polypoid with cup-shaped craters filled with parakeratotic cells that blended into keratinocytes of an acanthotic and papillomatous epidermis. There was a neutrophilic infiltrate of varying intensity between plump parakeratotic cells and keratinocytes, near the surface of the epidermis. Aggregates of foam cells were present in the papillary dermis, which was highly vascular. A plasma cell predominant infiltrate was seen at the base in a bandlike fashion. Despite the architectural resemblance of verruciform xanthoma to verrucous mucocutaneous lesions related to human papillomavirus infection, it was not detected by either immunohistochemistry, in situ hybridization, polymerase chain reaction, or Southern blot analysis in any case. The foam cells were weakly positive for cytokeratin and for Factor XIIIa but negative for S-100 protein. The KP1 and Mac 387 immunostain showed focal weak staining in foam cells. We postulate that a cascade of events pursue after initial keratinocytic damage attracting neutrophils, with subsequent phagocytosis of necrotic keratinocytic debris by dermal dendrocytes, eventually leading to the ultimate manifestation of the lesion as verruciform xanthoma. The etiologic agent remains elusive, but based on our findings, we conclude that verruciform xanthoma is most likely not a human papillomavirus-associated squamoproliferative lesion and that the foam cells, a histologic hallmark of the lesion, are most likely derived from dermal dendritic cells.
疣状黄色瘤是一种病因不明的罕见临床病理实体，主要发生于口腔黏膜。黏膜下基质或乳头真皮层中泡沫细胞聚集并伴有疣状上皮增生是该病变的特征。疣状黄色瘤发生于口腔外（皮肤）极为罕见，大多报道见于生殖器皮肤。本文研究了5例口腔外皮肤疣状黄色瘤（3例来自阴囊，1例来自阴茎，1例来自鼻部）及1例组织学“模拟物”（来自鼻部皮肤）。病变均为孤立性、隆起性或息肉样，有杯状凹陷，内充满不全角化细胞，这些细胞与棘层肥厚和乳头瘤样表皮的角质形成细胞相融合。在表皮表面附近，饱满的不全角化细胞与角质形成细胞之间有不同程度的中性粒细胞浸润。乳头真皮层有泡沫细胞聚集，此处血管丰富。在病变底部可见以带状分布为主的浆细胞浸润。尽管疣状黄色瘤在结构上与人类乳头瘤病毒感染相关的疣状黏膜皮肤病变相似，但在任何病例中，通过免疫组织化学、原位杂交、聚合酶链反应或Southern印迹分析均未检测到病毒。泡沫细胞对角蛋白和因子ⅩⅢa呈弱阳性，但对S-100蛋白呈阴性。KP1和Mac 387免疫染色显示泡沫细胞有局灶性弱阳性。我们推测，在初始角质形成细胞损伤后会引发一系列事件，吸引中性粒细胞，随后真皮树突状细胞吞噬坏死的角质形成细胞碎片，最终导致病变表现为疣状黄色瘤。病因仍不明确，但根据我们的研究结果，我们得出结论，疣状黄色瘤很可能不是人类乳头瘤病毒相关的鳞状上皮增生性病变，且病变的组织学特征性泡沫细胞很可能来源于真皮树突状细胞。
