日本未熟児新生児学会雑誌　第23巻第2号　59～64頁（2011年）

日本未熟児新生児学会雑誌　23(2)：269-274；2011　

受付日：平成22．07．01

受理日：平成23．01．31

巨大硬膜動静脈瘻に対しコイル塞栓術を施行した早産児例

Premature Infant Receiving Coil Embolism Due to Giant Dural Arteriovenous Fistula with Hydrops Fetalis

山形県立中央病院　小児科

Department of Pediatrics, Yamagata prefectural central hospital

佐藤裕子・饗場　智・閑野将行・渡辺　哲・内田俊彦・渡辺眞史

Hiroko SATO，Satoru AIBA，Masayuki KANNO，Satoshi WATANABE，Toshihiko UCHIDA，Masashi WATANABE

Key Words：dural arteriovenous fistula，coil embolism，premature infant，fetal hydrops，DIC

　症例は在胎30 週1 日，2,250g の男児。在胎28 週2 日，胎児超音波検査で頭蓋内に囊胞状病変の指摘を受け紹介された。胎児超音波カラードプラー検査で病変内の血流と皮下組織の肥厚，胸腹水を認めた。胎児MRI では外頸動脈から病変へ向かう流入血管が描出された。以上より巨大な硬膜動静脈瘻と診断した。両親に治療困難の可能性を説明したが治療の希望を示したので，在胎30 週1 日に緊急帝王切開で娩出された。生後150 分から43 個のGuglielmi Detachable Coil による流入動脈塞栓術を行い，動静脈瘻の血液量は減少した。全身浮腫と循環動態は改善したが，動静脈瘻内での播種性血管内凝固症候群（DIC）による2 回の脳室内出血と肺出血を合併し，日齢26に死亡した。胎児異常の早期発見と術後のDIC 対策が課題として残った。

　We reported a case of low birth weight boy infant（2,250g）with dural arteriovenous fistula born at 30.1 weeks of gestation. Fetus sonography showed a cystic lesion in the skull at 28 weeks of gestation, and mother was transferred to our hospital. Fetus ultrasound color flow Doppler showed bloodstream in the lesion, thickness of the subcutaneous tissue, and hydrothorax. Fetal magnetic resonance imaging（MRI）angiography revealed the blood vessel which flowed in the lesion from the external carotid arteries. Based on these findings, a diagnosis of giant dural arteriovenous fistula was made. To his parents, we explained his conditions including the possibility of intrauterine fetal death due to heart failure and the difficulty of recovery by treatments. The parents preferred active treatments, and emergency caesarean section was performed at 30.1 weeks of gestation. Apgar score was 3/3（1 minute/5 minutes）．One-hundred and fifty minutes after his birth, coil embolism to the inflow artery of dural arteriovenous fistula was performed. We used 43 GDC coils, and the bloodstream was remarkably decreased. After the embolism, anasarca was improved due to improvement of heart failure. However, disseminated intravascular coagulation（DIC）syndrome became advance, and complications of intraventricular and pulmonary hemorrhage were developed twice. He was died 26 days after the birth. This case showed the importance of early detection of fetus abnormalities, and a difficulty of controlling hemorrhage accompanied by DIC.