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Neurological Surgery No Shinkei Geka Volume 46, Issue 3 （March 2018）

Neurological Surgery 脳神経外科 46巻3号（2018年3月発行）

Japanese English

症例

破格側副血行路内の網状血管（twig-like networks）に起因すると考えられる脳出血で発症した内頚動脈終末部（C1）形成不全の1例 A Case of Segmental Dysgenesis at the Terminal Portion（C1）of the Internal Carotid Artery with an Anomalous Collateral Vessel Containing Plexiform（Twig-like）Networks as a Possible Cause of Intracerebral Hemorrhage 樋上翔大 ¹ , 大園恵介 ² , 牛島隆二郎 ² , 内田大貴 ^2,3 , 日宇健 ² , 川原一郎 ² , 小野智憲 ² , 戸田啓介 ^2,4 , 堤圭介 ² Shota HIGAMI ¹ , Keisuke OZONO ² , Ryujiro USHIJIMA ² , Daiki UCHIDA ^2,3 , Takeshi HIU ² , Ichiro KAWAHARA ² , Tomonori ONO ² , Keisuke TODA ^2,4 , Keisuke TSUTSUMI ² ¹独立行政法人国立病院機構長崎医療センター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聖隷浜松病院脳神経外科 ⁴独立行政法人国立病院機構長崎川棚医療センター脳神経外科 ¹Residency Program, National Hospital Organization Nagasaki Medical Center ²Department of Neurosurgery, National Hospital Organization Nagasaki Medical Center ³Department of Neurosurgery, Seirei Hamamatsu General Hospital ⁴Department of Neurosurgery, National Hospital Organization Nagasaki Kawatana Medical Center キーワード： internal carotid artery dysgenesis , terminal (C1) segment , anomalous collateral artery , primitive plexiform (twig-like) arterial networks , intracerebral hemorrhage Keyword: internal carotid artery dysgenesis , terminal (C1) segment , anomalous collateral artery , primitive plexiform (twig-like) arterial networks , intracerebral hemorrhage pp.235-245

発行日 2018年3月10日 Published Date 2018/3/10

DOI https://doi.org/10.11477/mf.1436203710

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Abstract 文献概要
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参考文献 Reference

Ⅰ．はじめに

　内頚動脈（internal carotid artery：ICA）形成不全は約200例集計されているが¹¹⁾，ICA終末部（C1）に限局する症例は極めて稀である⁶⁾．今回われわれは，側副血行路内の網状血管（twig-like networks）^{1,13,21,22,24)}が出血源と考えられる脳出血（intracerebral hemorrhage：ICH）で発症した，ICA-C1部形成不全の1例を経験した．特異的画像所見を提示するとともに，その発生過程や出血機序などについて，文献的考察を加えて報告する．

　A 65-year-old woman was transferred to our hospital because of loss of consciousness along with a right hemiplegia due to an intracerebral hematoma（ICH）that extended from the left temporal lobe to the lateral part of the basal ganglia. CT angiograms（CTA）and 3D-rotational angiograms showed an absence of the distal C1 segment of the left internal carotid artery（ICA), and an anomalous collateral vessel arising from the proximal end of the left A2 with plexiform（twig-like）arterial networks, supplying antegrade blood flow to the left middle cerebral artery territory. The caliber of the left carotid arterial system decreased from the origin to the terminal end. The source images of CTA revealed that the plexiform arteries were very close to the ICH, suggesting that the vessels might have caused the bleeding. Because a hypoplastic left carotid canal was observed, the segmental dysgenesis of the C1 portion of the left ICA seemed to have occurred during the early embryonic period. T2-weighted DRIVE images of MRI demonstrated the presence of a cord-like rudimentary structure corresponding to the distal C1 and proximal M1 portions. The patient was managed conservatively and was transferred to another hospital for subsequent rehabilitation.

　Segmental dysgenesis of the C1 portion is extremely rare. To our knowledge, only a single report has been published. The present findings suggest that 1）an anomalous collateral vessel corresponding to Heubner's artery evolved with residual primitive plexiform（twig-like）arterial networks and 2）this anomalous structure has not been proposed as a type of collateral systems for ICA dysgenesis.