Chan J K, Fletcher C D, Hicklin G A, Rosai J
Department of Pathology, Yale University School of Medicine, New Haven, Connecticut 06510.
Am J Surg Pathol. 1990 Nov;14(11):1036-46. doi: 10.1097/00000478-199011000-00005.
A histologically distinctive cutaneous hemangioma occurring in two patients with biopsy-proven multicentric Castleman's disease associated with POEMS (polyneuropathy, organomegaly, endocrinopathy, M-protein, skin changes) syndrome are reported. The lesions were multiple, and appeared as red to purple papules over the trunk and proximal limbs. Microscopically, ecstatic dermal vascular spaces were seen filled with aggregates of capillaries, resulting in structures reminiscent of renal glomeruli. Interspersed between the blood-filled capillary loops were plump "stromal" cells possessing clear vacuoles and periodic acid-schiff-positive eosinophilic globules. These cells had the immunohistochemical profile of endothelial cells (positivity for factor VIII-related antigen, and negativity for leukocyte common antigen, macrophage marker, and muscle-specific actin), and probably represented immature elements that had accumulated immunoglobulins and other proteinaceous material from the circulation. Because vascular lesions may appear before the full-blown POEMS syndrome develops, we suggest careful evaluation and follow-up of all patients presenting with glomeruloid hemangioma or cherry-type capillary hemangioma with focal glomeruloid features for potential development of this syndrome.
报告了两例经活检证实为多中心Castleman病并伴有POEMS(多发性神经病、器官肿大、内分泌病、M蛋白、皮肤改变)综合征的患者发生的一种组织学上独特的皮肤血管瘤。病变为多发,表现为躯干和近端肢体上的红色至紫色丘疹。显微镜下可见扩张的真皮血管腔隙内充满毛细血管聚集体,形成类似肾小球的结构。在充满血液的毛细血管袢之间散在着丰满的“基质”细胞,这些细胞具有清晰的空泡和过碘酸-希夫染色阳性的嗜酸性小球。这些细胞具有内皮细胞的免疫组化特征(对VIII因子相关抗原呈阳性,对白细胞共同抗原、巨噬细胞标志物和肌肉特异性肌动蛋白呈阴性),可能代表从循环中积累了免疫球蛋白和其他蛋白质物质的未成熟成分。由于血管病变可能在典型的POEMS综合征出现之前就已出现,我们建议对所有出现肾小球样血管瘤或具有局灶性肾小球样特征的樱桃型毛细血管血管瘤的患者进行仔细评估和随访,以监测该综合征的潜在发展。