日本新生児成育医学会雑誌　第31巻　第2号　152～157頁（2019年）

日本新生児成育医学会雑誌　31(2)：454-459；2019　

受付日：2018．05．22

受理日：2019．02．12

新生児期にKommerell 憩室による食道通過障害を来した血管輪の1 例

Esophageal Obstruction in A Neonate Caused by A Vascular Ring Involving Kommerell’s Diverticulum：A Case Report

＊ 1 亀田メディカルセンター　小児科．＊ 2 同　新生児科

＊ 1 Department of Pediatrics, Kameda Medical Center，＊ 2 Department of Neonatology, Kameda Medical Center

村上　楽＊ 1・水谷佳世＊ 2・中島隼也＊ 2・近藤　敦＊ 2・佐藤弘之＊ 2

Gaku MURAKAMI ＊ 1，Kayo MIZUTANI ＊ 2，Junya NAKAJIMA ＊ 2，Atsushi KONDO ＊ 2，Hiroyuki SATOH ＊ 2

Key Words：vascular ring，Kommerell’s diverticulum，esophageal symptom，neonate

　新生児期における血管輪の稀な初発症状として，食道通過障害を呈した症例を経験したので報告する。症例は在胎40 週0 日に経膣分娩で合併症なく2,892g で出生した男児である。日齢2 に他疾患を契機にNICU 入室となり，右側大動脈弓と左鎖骨下動脈起始異常，動脈管索にKommerell 憩室を伴う血管輪と診断された。入室後に食道通過障害による症状が気管狭窄症状に先行し，一時経管栄養も導入された。食道症状は生後6 週頃に自然軽快したが，それ以降に気管狭窄症状が出現し，8 か月時に根治手術を受け，その後の経過は良好である。新生児の嘔吐，哺乳困難，体重増加不良は，新生児科医・小児科医が遭遇する頻度の多い症状だが，その鑑別に血管輪も入れておく必要がある。また血管輪例では気管・食道それぞれの評価が肝要である。造影CT は必要不可欠な検査だが，食道の機能的な評価について限界がある。その点で食道造影は依然として有用である。

　Among conditions involving the presence of vascular ring in the neonatal period, tracheal stenosis is the most common initial presenting symptom, with initial esophageal compression rarely reported as the cause for presentation, while esophageal symptom is occasionally reported in infants and toddlers. Here, we describe a case of a male neonate born at 40 gestational weeks with a birth weight of 2,892g who was admitted to the neonatal intensive care unit for other reasons, but was later found to have partial esophageal obstruction caused by a vascular ring. The neonate had dysphagia and was diagnosed with esophageal compression caused by a vascular ring involving Kommerell’s diverticulum preceding tracheal symptoms. The vascular ring comprised a right-sided aortic arch, aberrant left subclavian artery, ligamentum arteriosum, and Kommerell’s diverticulum. Initial management involved tube feeding, with spontaneous resolution of esophageal symptoms at approximately 6 weeks. The child later developed tracheal stenosis and underwent successful radical surgical correction at the age of 8 months. Vomiting, feeding difficulties, and failure to thrive are common problems encountered in neonates by neonatologists and pediatricians, with vascular rings as a rare but important differential diagnosis. When vascular ring is suspected, evaluation of both the airway and the esophagus is important. Enhanced computed tomography（ CT） is an essential radiologic modality；however, it has limitations in the functional assessment of the esophagus. In such cases, esophagography retains an important role. Therefore, a combination of esophagography and enhanced CT is useful for the diagnosis.