Kaito Satoshi, Harada Sakiko, Tsukada Masao, Kojima Akira, Iwama Kanichi, Yamada Kazunari, Kajiwara Kouichi, Morikawa Kentaro, Shimizu Keiki, Kozai Yasuji, Koudo Hideki
Department of Hematology, Tokyo Metropolitan Tama Synthesis Medical Center.
Department of Emergency Rescue, Tokyo Metropolitan Tama Synthesis Medical Center.
Rinsho Ketsueki. 2018;59(1):45-50. doi: 10.11406/rinketsu.59.45.
A 35-year-old man who previously underwent splenectomy for hereditary spherocytosis at age 29 visited our hospital complaining of fatigue that had started 7 days ago and right upper abdominal pain. Laboratory data showed increased white blood cell and eosinophil count accompanied by severe transaminitis and clotting abnormalities. Computed tomography scan showed multiple embolisms in the portal vein, superior mesenteric vein, right pulmonary artery, and inferior vena cava. Severe liver damage presumably caused by portal vein thrombosis was also observed. Anticoagulant therapies consisting of continuous arterial infusion of urokinase from the superior mesenteric artery and an intravenous infusion of recombinant human thrombomodulin and heparin dissolved the systemic thrombosis. Concurrently administered prednisone decreased the eosinophil count. With regard to eosinophilia, we were unable to find any connective tissue diseases, antibodies to parasites, or genetic anomalies including PDGFRA, PDGFRB, and FGFR1. Hence we diagnosed the patient with idiopathic hypereosinophilic syndrome (HES). Although thromboembolisms in patients with HES have been reported, the literature on portal vein thrombosis associated with HES is scarce. In the present case, the previous splenectomy may have contributed to the portal vein thrombosis.
一名35岁男性,29岁时因遗传性球形红细胞增多症接受了脾切除术,因7天前开始出现疲劳及右上腹疼痛前来我院就诊。实验室检查数据显示白细胞和嗜酸性粒细胞计数升高,伴有严重的转氨酶升高和凝血异常。计算机断层扫描显示门静脉、肠系膜上静脉、右肺动脉和下腔静脉多处栓塞。还观察到可能由门静脉血栓形成导致的严重肝损伤。由肠系膜上动脉持续动脉输注尿激酶以及静脉输注重组人血栓调节蛋白和肝素组成的抗凝治疗溶解了全身血栓。同时给予的泼尼松降低了嗜酸性粒细胞计数。关于嗜酸性粒细胞增多,我们未发现任何结缔组织病、寄生虫抗体或包括血小板衍生生长因子受体α(PDGFRA)、血小板衍生生长因子受体β(PDGFRB)和纤维母细胞生长因子受体1(FGFR1)在内的基因异常。因此,我们诊断该患者患有特发性高嗜酸性粒细胞综合征(HES)。虽然已有报道HES患者出现血栓栓塞,但与HES相关的门静脉血栓形成的文献很少。在本病例中,既往的脾切除术可能促成了门静脉血栓形成。