日本新生児成育医学会雑誌　35(2)：280-285；2023　

日本新生児成育医学会雑誌　第35巻　第2号　131 ～136頁（2023年）

受付日：2022．10．20

受理日：2023．02．27

動静脈奇形との鑑別に苦慮し病理所見から診断した先天性血管腫の新生児例

A Neonatal Case of Congenital Hemangioma Diagnosed Based on Pathological Findings after Difficulty Differentiating It from Arteriovenous Malformation

＊ 1 奈良県立医科大学附属病院　小児科学，＊ 2 奈良県総合医療センター　新生児集中治療部

＊ 1 Department of Pediatrics, Nara Medical University Hospital， ＊ 2 Department of Neonatal Intensive Care Unit, Nara Prefecture General Medical Center

蜂須賀宗嗣＊ 1 ＊ 2・桐村章大＊ 2・安原　肇＊ 2・恵美須礼子＊ 2・扇谷綾子＊ 2・箕輪秀樹＊ 2

Soshi HACHISUKA ＊ 1 ＊ 2，Akihiro KIRIMURA ＊ 2，Hajime YASUHARA ＊ 2，Reiko EBISU ＊ 2，Ayako OHGITANI ＊ 2，Hideki MINOWA ＊ 2

Key Words：neonate，congenital hemangioma，arteriovenous malformation，high-output heart failure

動静脈奇形との鑑別に苦慮し，生検による病理所見から先天性血管腫と診断した症例を経験したので報告する。 症例は日齢1 の男児。在胎39 週3 日，出生体重3,158g。出生時から左側頸部から背部にかけて血管腫様の皮膚病変 を認め，頭頸部MRI/MRA および造影CT 検査により動静脈奇形が疑われた。生後より頸部病変のシャント血流増 大に伴う高拍出性心不全により呼吸障害を認めたが，徐々に改善し日齢39 に退院した。外表所見や画像所見では確 定診断がつかず治療方針を明確にするために生検を行った。病理所見では小型の脈管が集簇する領域と近接して静 脈様の拡張脈管を認めた。免疫組織化学染色では血管内皮のanti-GLUT1 抗体は陰性であり先天性血管腫と診断し た。生後1 歳1 か月現在，心不全徴候はなく頸部病変の有意な変化は認めていない。先天性血管腫と動静脈奇形は 臨床所見が類似しており鑑別に苦慮するが，治療方針が異なるため生検による病理診断を要する症例が存在する。

We report a case of congenital hemangioma diagnosed based on pathological findings on biopsy, despite the presence of arteriovenous malformation. The patient was a one-day-old boy. He was born by transvaginal labor at a gestational age of 39 weeks and 3 days and weighed 3,158 g. Hemangioma-like skin lesions were observed from birth on the left side of the neck to the back, and arteriovenous malformation was suspected from MRI/MRA and contrast-enhanced CT scan. From birth, respiratory distress was observed due to hyperpulsatile heart failure associated with increased shunt blood flow in the cervical lesion, but the patient gradually improved and was discharged at 39 days of age. Since no definitive diagnosis could be made based on the external and imaging findings, a biopsy was performed to clarify the treatment plan. Pathological findings showed areas of small vascular conglomerations and a venous-like dilated vasculature in close proximity to the normal vasculature. Immunohistochemical staining of the vascular endothelium with anti-GLUT1 antibody was negative. A diagnosis of congenital hemangioma was made. At 1 year and 1 month of age, there were no signs of heart failure and no significant changes in the neck lesions. Although it is difficult to distinguish congenital hemangioma from arteriovenous malformation because of the similar clinical findings, there are cases in which pathological diagnosis by biopsy is required because different treatment strategies are needed.