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 35(1): 38-42 (2019)
doi:10.9794/jspccs.35.38

症例報告Case Report

急性期川崎病診療のピットフォール左冠動脈肺動脈起始症Pitfall in Acute Care of Kawasaki Disease: Anomalous Origin of the Left Coronary Artery from the Pulmonary Artery

久留米大学医学部小児科学教室Department of Pediatrics and Child Health, Kurume University School of Medicine ◇ Fukuoka, Japan

受付日:2018年4月16日Received: April 16, 2018
受理日:2018年11月8日Accepted: November 8, 2018
発行日:2019年3月1日Published: March 1, 2019
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川崎病は日常診療で遭遇する機会が多く,合併症として冠動脈拡大・瘤の診断は重要である.一方,冠動脈拡大・瘤を示す疾患には,左冠動脈肺動脈起始症,冠動脈瘻,左冠動脈閉鎖症などの先天性冠動脈異常もある.症例は6歳女児で,歳時に川崎病に罹患し,前医に入院した.免疫グロブリン大量療法を施行され,速やかに解熱したが,初回心エコー図検査時より右冠動脈拡大があり,縮小することなく残存した.川崎病性右冠動脈拡大,軽症僧帽弁閉鎖不全,動脈管開存症疑いとして前医で外来管理されていた.5歳時より全力疾走時に胸痛を訴えるようになった.運動負荷心電図検査を施行したところ,自覚症状はなかったが,V3–6誘導で0.1–0.2 mVの下降型ST低下が認められ,精査加療目的で当院紹介された.当院で冠動脈造影CT検査を施行し,左冠動脈肺動脈起始症と確定診断した.術前の精査では左室前側壁領域の軽度壁運動低下を認め,左冠動脈移植手術を行った.今回,川崎病性右冠動脈拡大と診断され,無治療経過観察されていた左冠動脈肺動脈起始症の1小児例を経験した.急性期川崎病診療のピットフォールとして,注意喚起すべきと考えたので報告する.

Kawasaki disease (KD) is the most prevalent acquired vasculitis in children; coronary artery dilation and aneurysms are the most important complications of this disease. Alternatively, coronary artery dilation can be observed in rare congenital coronary artery anomalies, such as anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA). At the age of 3, the patient suffered from typical KD, which was successfully treated with immunoglobulin infusion; however, right coronary dilation was noted at 4 days of illness. She was followed-up for the persistent right coronary artery dilation. At the age of 6, she complained of chest pain, and exercise stress test revealed ST segment depression in leads V3–V6, which resulted in the tentative diagnosis of coronary artery fistula. She was referred to our hospital for further evaluation and treatment. X-ray coronary computed tomography lead to the diagnosis of ALCAPA and stress myocardial scintigraphy confirmed ischemia of the anterolateral wall of the left ventricle; therefore, she underwent successful direct reimplantation of the anomalous left coronary artery to the aortic root. Here, we report the case of a patient with ALCAPA who was misdiagnosed as having coronary artery dilation associated with KD, and the patient was followed-up without appropriate treatment. We found that misdiagnosis of congenital anomalies can be a drawback of echocardiographic evaluation of coronary arteries in patients with acute KD, and clinicians must be able to properly distinguish between both the plausible causes of dilation.

Key words: coronary artery aneurysm; anomalous origin of the left coronary artery from the pulmonary artery; Kawasaki disease; echocardiography

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This page was last modified on 2019-03-15T15:23:49.000+09:00


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