Dunphy C H, Sotelo-Avila C, Luisiri A, Chu J Y
Department of Pathology, St Louis University School of Medicine, MO 63104.
Arch Pathol Lab Med. 1994 Aug;118(8):837-9.
Paroxysmal nocturnal hemoglobinuria is an acquired clonal expansion of bone marrow stem cells that are deficient in the decay-accelerating factor, which is a complement regulatory glycoprotein (CD55), as well as in the membrane inhibitor of reactive lysis (CD59) and the C8-binding protein. These proteins are deficient on the membranes of red blood cells, granulocytes, monocytes, and platelets. The disorder is associated with intermittent hemolytic anemia, hemoglobinuria, infection, a tendency toward bone marrow aplasia, and venous thromboses. The thromboses, on resolution, may give rise to endothelial proliferation that may cause ischemia and ulceration, or, alternatively, the thromboses may cause ulceration leading to a granulation tissue response with exaggerated endothelial proliferation. We report a second case of paroxysmal nocturnal hemoglobinuria that presented roentgenographically as an ulcerated circumferential duodenal mass secondary to venous thrombosis accompanied by florid papillary endothelial hyperplasia. We also review the literature concerning this phenomenon.
阵发性睡眠性血红蛋白尿是一种获得性骨髓干细胞克隆性扩增疾病,这些干细胞缺乏衰变加速因子,该因子是一种补体调节糖蛋白(CD55),同时还缺乏反应性溶解膜抑制剂(CD59)和C8结合蛋白。这些蛋白质在红细胞、粒细胞、单核细胞和血小板膜上缺乏。该疾病与间歇性溶血性贫血、血红蛋白尿、感染、骨髓再生障碍倾向和静脉血栓形成有关。血栓溶解后,可能会引起内皮细胞增殖,进而导致局部缺血和溃疡形成;或者,血栓可能导致溃疡,引发肉芽组织反应,并伴有内皮细胞过度增殖。我们报告了第二例阵发性睡眠性血红蛋白尿病例,其X线表现为十二指肠环周溃疡性肿块,继发于静脉血栓形成,并伴有明显的乳头状内皮细胞增生。我们还回顾了有关这一现象的文献。