Department of Hematology, Latvian Oncology Centre, Riga, Latvia.
Riga Stradins University, Riga, Latvia.
Am J Case Rep. 2022 Mar 20;23:e934287. doi: 10.12659/AJCR.934287.
BACKGROUND Classical hemophilia, or hemophilia A, is an X-linked recessive genetic disorder characterized by deficiency in clotting factor VIII. Renal artery aneurysms (RAAs) are also rare and are defined as a focal dilatation of the renal artery that exceeds 1.5 cm in diameter. These 2 rare conditions - giant RAA and hemophilia A - were simultaneously observed in our patient. This report presents a male patient with hemophilia A with a 10-cm aneurysm of the right renal artery, which was treated with transarterial coil embolization and factor VIII infusion. The giant RAA was an incidental finding and was suspected after the abdominal ultrasound (US). CASE REPORT We present the case of a 10-cm right RAA in a 54-year-old man with hemophilia A. The patient had a congenital severe coagulation factor VIII deficiency (hemophilia A). He presented at a routine hematologist visit with an atypical symptom of severe symmetrical leg edema. Laboratory tests showed increased levels of creatinine and proteinuria. Investigations proceeded with computed tomography (CT) and digital subtraction angiography (DSA). Endovascular coiling of the aneurysm was performed with perioperative recombinant coagulation factor VIII substitution, and the recovery was uneventful. At 6-year follow-up there are no signs of proteinuria, and kidney function was stable. CONCLUSIONS We present a case of renal artery aneurysm effectively treated by endovascular embolization, showing the importance of managing patients with hemophilia A according to a guidelines-based multidisciplinary approach and ensuring the lowest possible risk of peri- and intraoperative complications by using minimally-invasive treatments.
经典血友病,又称血友病 A,是一种 X 连锁隐性遗传疾病,其特征是凝血因子 VIII 缺乏。肾动脉动脉瘤(RAAs)也很少见,定义为肾动脉的局灶性扩张,直径超过 1.5 厘米。这两种罕见的情况 - 巨大的 RAA 和血友病 A - 在我们的患者中同时观察到。本报告介绍了一名男性血友病 A 患者,其右侧肾动脉有一个 10 厘米的动脉瘤,通过经动脉线圈栓塞和 VIII 因子输注进行治疗。巨大的 RAA 是偶然发现的,在腹部超声(US)检查后怀疑是这种疾病。
我们介绍了一名 54 岁男性患者的右侧肾动脉 10 厘米巨大 RAA,该患者患有先天性严重凝血因子 VIII 缺乏症(血友病 A)。他因严重对称性腿部水肿的非典型症状在常规血液科就诊。实验室检查显示肌酐和蛋白尿水平升高。检查进行了计算机断层扫描(CT)和数字减影血管造影(DSA)。在围手术期用重组凝血因子 VIII 替代物进行了动脉瘤的血管内线圈栓塞,恢复顺利。6 年随访时无蛋白尿迹象,肾功能稳定。
我们报告了一例肾动脉动脉瘤通过血管内栓塞有效治疗的病例,表明根据基于指南的多学科方法管理血友病 A 患者并通过使用微创治疗尽可能降低围手术期并发症风险的重要性。