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Neurological Surgery No Shinkei Geka Volume 47, Issue 10 （October 2019）

Neurological Surgery 脳神経外科 47巻10号（2019年10月発行）

Japanese English

症例

皮質性くも膜下出血に近接する急性期微小皮質梗塞を認めた可逆性脳血管攣縮症候群の1例 Reversible Cerebral Vasoconstriction Syndrome with Cortical Subarachnoid Hemorrhage due to Acute Cortical Infarction Beneath the Sulcus:A Case Report 江端由穂 ¹ , 福田雄高 ^2,3 , 中村光流 ^1,2 , 近松元気 ^1,2 , 塩崎絵理 ^1,2 , 森塚倫也 ² , 日宇健 ² , 川原一郎 ² , 小野智憲 ² , 原口渉 ² , 牛島隆二郎 ² , 堤圭介 ² Yuho EBATA ¹ , Yutaka FUKUDA ^2,3 , Hikaru NAKAMURA ^1,2 , Genki CHIKAMATSU ^1,2 , Eri SHIOZAKI ^1,2 , Tomoya MORITSUKA ² , Takeshi HIU ² , Ichiro KAWAHARA ² , Tomonori ONO ² , Wataru HARAGUCHI ² , Ryujiro USHIJIMA ² , Keisuke TSUTSUMI ² ¹独立行政法人国立病院機構長崎医療センター脳神経外科初期研修プログラム ²独立行政法人国立病院機構長崎医療センター脳神経外科 ³福田脳神経外科病院 ¹Residency Program, Department of Neurosurgery, National Hospital Organization Nagasaki Medical Center ²Department of Neurosurgery, National Hospital Organization Nagasaki Medical Center ³Fukuda Neurosurgical Hospital キーワード： reversible cerebral vasoconstriction syndrome , RCVS , cortical subarachnoid hemorrhage , cSAH , acute cortical infarction , hemorrhagic infarction , ischemia-reperfusion injury Keyword: reversible cerebral vasoconstriction syndrome , RCVS , cortical subarachnoid hemorrhage , cSAH , acute cortical infarction , hemorrhagic infarction , ischemia-reperfusion injury pp.1073-1079

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

DOI https://doi.org/10.11477/mf.1436204076

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Abstract 文献概要
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Ⅰ．はじめに

　可逆性脳血管攣縮症候群（reversible cerebral vasoconstriction syndrome：RCVS）に合併する皮質性くも膜下出血（cortical subarachnoid hemorrhage：cSAH）は発症早期に約1/3の症例で認められるが，その出血機序については明らかではない^3,7)．今回われわれは，雷鳴様頭痛（thunderclap headache：TH）発症後2日目の画像検査でcSAHを認め，血腫が最も厚い脳溝部に近接して急性期微小皮質梗塞を合併したと考えられるRCVSの1例を経験した．稀な症例であり，RCVSに伴うcSAHの臨床像やその発生機序について，文献的考察を加えて報告する．

　We report a rare case of reversible cerebral vasoconstriction syndrome（RCVS）with cortical subarachnoid hemorrhage（cSAH）associated with a fresh cortical infarction beneath the sulcus with thick cSAH.

　A 34-year-old woman presented with history of thunderclap headache. She was transferred to our hospital for further examination of a cSAH in the left frontal lobe. Results of the cerebrospinal fluid examination were unremarkable, but three-dimensional rotational angiography revealed multiple instances of narrowing of the cortical branches of the anterior and middle cerebral arteries, suggesting the diagnosis of RCVS. Diffusion weighted imaging（DWI）demonstrated a small cortical area with high-signal intensity around the sulcus , where a thick cSAH clot was observed. This cortical lesion appeared as low-signal intensity on the apparent diffusion coefficient maps, and the follow-up T2-weighted images（obtained 3 months after onset）demonstrated a residual lesion that was smaller than the initial DWI abnormality with high-signal intensity;thus indicating the presence of a coincident fresh cortical infarction. The position of the infarct next to the thickest portion of cSAH suggested that it was the bleeding source of the cSAH. Ten days after onset, the cerebral blood flow and volume in the cortex around the cSAH increased as compared to the same area on the contralateral side.

　These findings suggested that at least one of the bleeding mechanisms of the cSAH was related to the hemorrhagic infarction or subpial hemorrhage resulting from the “ischemia-reperfusion injury” due to the acute disturbance of the pial vessel microcirculation with subsequent rapid resolution of the blood flow during the early phases of RCVS. These dynamics could not be demonstrated with contemporary angiographic imaging.