Sakai Naoto, Akamine Soichi, Tokuyama Tsutomu, Sugiyama Kenji, Kanayama Naohiro, Namba Hiroki
Department of Neurosurgery, Hamamatsu University School of Medicine, Shizuoka.
Neurol Med Chir (Tokyo). 2011;51(11):780-3. doi: 10.2176/nmc.51.780.
A 37-year-old woman with congenital afibrinogenemia presented with chronic subdural hematoma (CSDH) manifesting as severe headache, nausea, and somnolence after a minor head trauma. Brain computed tomography scans showed a right subdural hematoma associated with midline shift. Laboratory studies showed prolongation of prothrombin time, activated partial thromboplastin time, and undetectably low level of fibrinogen. Until the present episode, she had received plasma-derived fibrinogen concentrate around menstruation and pregnancy. She had also suffered from spinal cord infarction due to vertebral artery occlusion. Burr-hole evacuation and drainage of CSDH was successfully performed using fibrinogen concentrate. The development of CSDH with afibrinogenemia is very rare. Although the past repeated administrations of fibrinogen concentrate were suspected to generate CSDH, paradoxical thrombotic complications caused by upregulation of prothrombin activation, thrombin generation, and growth factors released from platelets might be related to the development of CSDH with congenital afibrinogenemia.
一名37岁先天性无纤维蛋白原血症女性患者,在轻微头部外伤后出现慢性硬膜下血肿(CSDH),表现为严重头痛、恶心和嗜睡。脑部计算机断层扫描显示右侧硬膜下血肿伴中线移位。实验室检查显示凝血酶原时间、活化部分凝血活酶时间延长,纤维蛋白原水平低至检测不到。在本次发病前,她在月经和怀孕期间接受过血浆源性纤维蛋白原浓缩物治疗。她还曾因椎动脉闭塞发生过脊髓梗死。使用纤维蛋白原浓缩物成功进行了CSDH钻孔引流术。无纤维蛋白原血症合并CSDH的情况非常罕见。尽管过去多次使用纤维蛋白原浓缩物被怀疑会引发CSDH,但由凝血酶原激活上调、凝血酶生成以及血小板释放的生长因子引起的矛盾性血栓并发症可能与先天性无纤维蛋白原血症合并CSDH的发生有关。