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Neurological Surgery No Shinkei Geka Volume 47, Issue 8 （August 2019）

Neurological Surgery 脳神経外科 47巻8号（2019年8月発行）

Japanese English

症例

Primary intraventricular hemorrhageで発症した上矢状洞部硬膜動静脈廔の1例 A Case of Dural Arteriovenous Fistula Involving the Superior Sagittal Sinus that Presented as a Primary Intraventricular Hemorrhage 大園恵介 ^1,3 , 近松元気 ^1,2 , 中村光流 ^1,2 , 塩崎絵理 ^1,2 , 日宇健 ¹ , 堀江信貴 ³ , 川原一郎 ¹ , 小野智憲 ¹ , 原口渉 ¹ , 牛島隆二郎 ¹ , 堤圭介 ¹ Keisuke OZONO ^1,3 , Genki CHIKAMATSU ^1,2 , Hikaru NAKAMURA ^1,2 , Eri SHIOZAKI ^1,2 , Takeshi HIU ¹ , Nobutaka HORIE ³ , Ichiro KAWAHARA ¹ , Tomonori ONO ¹ , Wataru HARAGUCHI ¹ , Ryujiro USHIJIMA ¹ , Keisuke TSUTSUMI ¹ ¹国立病院機構長崎医療センター脳神経外科 ²国立病院機構長崎医療センター初期研修プログラム ³長崎大学脳神経外科 ¹Department of Neurosurgery, National Hospital Organization Nagasaki Medical Center ²Residency Program, National Hospital Organization, Nagasaki Medical Center ³Department of Neurosurgery, Nagasaki University school of Medicine キーワード： primary（isolated）intraventricular hemorrhage , deep medullary vein , dural arteriovenous fistula , superior sagittal sinus , varix（venous aneurysm） Keyword: primary（isolated）intraventricular hemorrhage , deep medullary vein , dural arteriovenous fistula , superior sagittal sinus , varix（venous aneurysm） pp.859-867

発行日 2019年8月10日 Published Date 2019/8/10

DOI https://doi.org/10.11477/mf.1436204037

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Ⅰ．はじめに

　硬膜動静脈廔（dural arteriovenous fistula：DAVF）が純粋な脳室内出血（primary intraventricular hemorrhage：PIVH）で発症することは稀であり^3,7)，報告例の中では横-S状静脈洞部（transvers-sigmoid sinus：TSS）DAVFが多くを占める²⁾．今回われわれは，PIVHで発症した上矢状洞部（superior sagittal sinus：SSS）DAVFの極めて稀な1例を経験したので，文献的考察を加えて報告する．

　We report an extremely rare case of dural arteriovenous fistula（DAVF）involving the superior sagittal sinus（SSS）that presented as a primary intraventricular hemorrhage（PIVH).

　A 79-year-old man who presented with disturbance of consciousness and vomiting was transferred to our hospital. The initial head CT revealed an intraventricular hemorrhage in the lateral, third, and fourth ventricles, predominantly involving the right lateral ventricle associated with obstructive hydrocephalus. CT angiography and three-dimensional rotational angiography（3D-RA）revealed SSS-DAVF with retrograde venous drainage from the cortical venous system to the subependymal venous system through engorged superficial/anastomotic/deep medullary or transcerebral veins, showing a pseudophlebitic pattern. A severe stenosis of the SSS was observed at the site just proximal to the shunting point. A varix of the right transvers caudate vein（TCV）was observed within the thickest portion of the hematoma in close contact with the wall of the right lateral ventricle. A stenosis of the branching point of the right TCV was detected on the 3D-RA image. The DAVF was completely cured after transarterial embolization. The varix gradually shrank and finally disappeared after 2 months.

　This is the first report of a case of PIVH due to SSS-DAVF with a subependymal varix considered as the bleeding point. Serial gadolinium-enhanced MR images clearly showed the shrinking of the varix. Our case is the second reported case of spontaneous resolution of the varix after embolization of the DAVF.

　The obstructive changes of the two venous outflow pathways（SSS/TCV）might further worsen the state of venous congestion in the deep medullary venous system and eventually lead to formation and rupture of the varix.