Ozaki S, Kawachi Y, Igaki T, Ogasawara N, Uchida T, Mori M, Setsu S
Department of Internal Medicine, Takamatsu Red Cross Hospital.
Rinsho Ketsueki. 1991 Nov;32(11):1509-14.
A 62-year-old woman was admitted to our hospital because of general malaise in May, 1987. No hepatosplenomegary, skin eruption or lymphadenopathy was detected. Laboratory examinations showed mild anemia, thrombocytopenia, normal leukocyte count with no lymphocyte abnormality, hypogranular neutrophils, elevated serum lactic dehydrogenase, increased C-reactive protein and hypoxia. Bone marrow aspirate was normocellular with dysplastic changes in erythroid and megakaryocytic lines which agree with a diagnosis of myelodysplastic syndrome (MDS). She was treated with prednisolone which relieved her symptom but she developed high fever, hemiplegia and disturbance of consciousness and died in August, 1987. Necropsy of the kidney revealed large mononuclear cells within the lumen of small blood vessels. Immunohistochemical study of these malignant cells showed positive reaction to the anti-LCA and anti-L26 antibodies. And electron microscopy showed no azure granules in these cells. Then we diagnosed as neoplastic angioendotheliosis (NAE). To our knowledge, this is the first report of NAE with abnormalities in myeloid, erythroid and megakaryocytic lineages. These results suggest that NAE with MDS originate from a multipotent stem cell.
一名62岁女性因全身不适于1987年5月入住我院。未发现肝脾肿大、皮疹或淋巴结病。实验室检查显示轻度贫血、血小板减少、白细胞计数正常且无淋巴细胞异常、中性粒细胞颗粒减少、血清乳酸脱氢酶升高、C反应蛋白增加和低氧血症。骨髓穿刺显示细胞数量正常,红系和巨核系有发育异常改变,符合骨髓增生异常综合征(MDS)的诊断。她接受了泼尼松龙治疗,症状得到缓解,但随后出现高热、偏瘫和意识障碍,并于1987年8月死亡。肾脏尸检显示小血管腔内有大的单核细胞。对这些恶性细胞的免疫组织化学研究显示,其对抗LCA和抗L26抗体呈阳性反应。电子显微镜检查显示这些细胞内无天青颗粒。于是我们诊断为肿瘤性血管内皮病(NAE)。据我们所知,这是首例伴有髓系、红系和巨核系异常的NAE报告。这些结果表明,伴有MDS的NAE起源于多能干细胞。