Molenaar W M, Rosman J B, Donker A J, Houthoff H J
Pathol Res Pract. 1987 Feb;182(1):98-106. doi: 10.1016/S0344-0338(87)80148-6.
The morphology and immunohistology in a case of malignant atrophic papulosis (Degos' disease), a rare vascular disorder of unknown etiology, are described. The vascular lesions affected middle class and small arteries and veins throughout the body and were histologically characterized by intimal proliferation in the absence of any appreciable inflammation. The lesions were categorized as early, intermediate or late. Early lesions consisted of cellular proliferation and edema of the intima with signs of immune complex deposition (IgM, C3). Thrombosis was occasionally present as a secondary phenomenon in the affected vessel segments. In intermediate lesions the edema decreased and smooth muscle proliferation became apparent. Late lesions consisted of acellular intimal sclerosis with hyalinization and narrowing or obliteration of the vascular lumen. The media of the vessels remained always intact. In comparing these features to the pathology and pathogenesis of other vascular disorders they resembled the vascular lesions in a murine model of lupus erythematodes in which also considerable intimal proliferation occurred with thrombotic occlusion, but without appreciable inflammation. The murine model is associated with sustained low levels of circulating immune complexes and it is tempting to assume the same for Degos' disease. The notion of an immune complex mediated non-inflammatory condition underlying this severe and often fatal vascular disorder of mainly young males may contribute to the eventual finding of a successful therapeutical regimen.
本文描述了恶性萎缩性丘疹病(德戈斯病)这一病因不明的罕见血管疾病的形态学和免疫组织学特征。血管病变累及全身的中小动脉和静脉,组织学特征为内膜增生且无明显炎症。病变分为早期、中期或晚期。早期病变表现为内膜细胞增殖和水肿,并伴有免疫复合物沉积(IgM、C3)迹象。血栓形成偶尔作为受累血管段的继发现象出现。中期病变中水肿减轻,平滑肌增殖明显。晚期病变由无细胞内膜硬化伴玻璃样变以及血管腔狭窄或闭塞组成。血管中层始终保持完整。将这些特征与其他血管疾病的病理和发病机制进行比较时,它们类似于红斑狼疮小鼠模型中的血管病变,该模型中也出现了相当程度的内膜增生伴血栓性闭塞,但无明显炎症。该小鼠模型与循环免疫复合物持续低水平有关,因此推测德戈斯病也存在同样情况。这种主要发生在年轻男性身上的严重且往往致命的血管疾病,其潜在病因是免疫复合物介导的非炎症状态,这一观点可能有助于最终找到成功的治疗方案。