日本小児循環器学会雑誌 Pediatric Cardiology and Cardiac Surgery

Online ISSN: 2187-2988 Print ISSN: 0911-1794
特定非営利活動法人日本小児循環器学会 Japanese Society of Pediatric Cardiology and Cardiac Surgery
〒162-0801東京都新宿区山吹町358-5アカデミーセンター Japanese Society of Pediatric Cardiology and Cardiac Surgery Academy Center, 358-5 Yamabuki-cho, Shinju-ku, Tokyo 162-0801, Japan
Pediatric Cardiology and Cardiac Surgery 32(6): 518-523 (2016)
doi:10.9794/jspccs.32.518

症例報告Case Report

経皮的経カテーテル動脈管閉鎖術を契機に急性発症し診断に至った左冠動脈肺動脈起始症の1例A Case of Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery Developed Acute Heart Failure by Transcatheter Ductus Arteriosus Closure

1JCHO中京病院中京こどもハートセンター小児循環器科Department of Pediatric Cardiology, Chukyo Children Heart Center, Japan Community Healthcare Organization Chukyo Hospital ◇ Aichi, Japan

2JCHO中京病院中京こどもハートセンター心臓血管外科Department of Cardiovascular Surgery, Chukyo Children Heart Center, Japan Community Healthcare Organization Chukyo Hospital ◇ Aichi, Japan

受付日:2016年9月1日Received: September 1, 2016
受理日:2016年11月4日Accepted: November 4, 2016
発行日:2016年11月1日Published: November 1, 2016
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10か月健診時の心雑音を契機に動脈管開存症(PDA),肺高血圧症(PH)と診断されて当院を受診した.12か月時に当院にて経皮的経カテーテル動脈管閉鎖術を施行したところ経胸壁心臓超音波検査にて心機能低下,血液生化学検査にて心筋逸脱酵素の上昇,心電図にて異常Q波が出現した.経胸壁心臓超音波検査再検にて主肺動脈からの左肺動脈起始を確認し左冠動脈肺動脈起始症(ALCAPA)と診断し,開胸手術を行った.ALCAPAは典型的には生後8週頃に肺血圧の低下に伴い左冠動脈の潅流が不良となり心不全を発症する疾患であるが,動脈管開存症(PDA)をはじめとした肺血流増多疾患を合併した場合,左冠動脈の潅流が維持されるため乳児期の心不全発症を免れうる.このためPDAを合併したALCAPAは動脈管閉鎖前の診断が極めて困難であるが,未診断下に動脈管閉鎖術を施行した場合に急激に左冠動脈潅流が悪化して心不全を発症することがある.PH合併PDAの症例では,経皮的経カテーテル動脈管閉鎖術前に冠動脈奇形も念頭に経胸壁超音波検査を行い,それでも否定できない場合は閉鎖前に左室造影や上行大動脈造影を考慮するべきである.

A 12-month-old boy who was diagnosed with a large patent ductus arteriosus (PDA) and pulmonary hypertension (PH) in another hospital was admitted to Chukyo Children Heart Center for catheter intervention. After transcatheter PDA closure, he developed myocardial dysfunction accompanied by elevated cardiac enzyme levels and an abnormal Q-wave on electrocardiography. Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) was detected on follow-up transthoracic echocardiography, and open chest surgery was performed. ALCAPA typically manifests in infancy with signs and symptoms of heart failure that are secondary to ischemic cardiomyopathy. However, it is very difficult to diagnose when additional congenital heart disease, for example, PDA, is present, because left coronary perfusion is maintained by PH. If PDA is treated without the diagnosis of ALCAPA, sudden deterioration may occur. We concluded that we should confirm the origin of the coronary artery using left ventriculography or aortography before transcatheter PDA closure if we cannot rule out the possibility of coronary artery anomalies by careful transthoracic echocardiography.

Key words: anomalous origin of the left coronary artery from the pulmonary artery; patent ductus arteriosus; acute myocardial infarction; catheter intervention; preoperative diagnosis

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