Department of Neurosurgery, Fujita Health University, Toyoake, Japan; Department of Neurosurgery, TOYOTA Memorial Hospital, Toyota, Japan.
Department of Neurosurgery, Fujita Health University, Toyoake, Japan; Department of Neurosurgery, Fujita Health University Okazaki Medical Center, Okazaki, Japan.
Clin Neurol Neurosurg. 2021 May;204:106612. doi: 10.1016/j.clineuro.2021.106612. Epub 2021 Mar 24.
Pial arteriovenous fistulas (AVFs) are rare vascular lesions; their exact pathophysiology is largely unknown. Pial AVFs have been reported to develop within capillary malformation-arteriovenous malformation (CM-AVM); however, only a few cases have been reported. Variants in the RASA1 gene have been reported as a cause of CM-AVM. We report the case of an adult patient with pial AVF, who carried variants in the RASA1 and COL4A2 genes. The patient in the current report was likely to have been affected by CM-AVM and the RASA1 variant seemed to be the primary factor in the pathogenesis of pial AVF. However, COL4A2 may have also contributed to the development of pial AVF because the COL4A2 and RASA1 variants have a common pathophysiology, wherein the patient develops lesions due to collagen type IV deficiency.
脑表面动静脉瘘(AVF)是一种罕见的血管病变;其确切的病理生理学在很大程度上尚不清楚。脑表面 AVF 已被报道在毛细血管畸形-动静脉畸形(CM-AVM)内发展;然而,仅有少数病例被报道。RASA1 基因的变异已被报道为 CM-AVM 的病因。我们报告了一例脑表面 AVF 的成年患者,该患者携带 RASA1 和 COL4A2 基因的变异。本报告中的患者可能患有 CM-AVM,而 RASA1 变异似乎是脑表面 AVF 发病机制的主要因素。然而,COL4A2 也可能促成了脑表面 AVF 的发展,因为 COL4A2 和 RASA1 变异具有共同的病理生理学,患者由于 IV 型胶原缺乏而发生病变。