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
Pediatric Cardiology and Cardiac Surgery 37(1): 57-63 (2021)

症例報告Case Report

modified Blalock–Taussig shunt感染性閉塞症例の臨床像Clinical Features of Infective Modified Blalock–Taussig Shunt Obstruction

あいち小児保健医療総合センター 小児心臓病センター 循環器科Department of Pediatric Cardiology, Kids’ Heart Center, Aichi Children’s Health and Medical Center ◇ Aichi, Japan

受付日:2020年7月2日Received: July 2, 2020
受理日:2020年10月6日Accepted: October 6, 2020
発行日:2021年4月1日Published: April 1, 2021

modified Blalock–Taussig shunt (mBTS)感染性閉塞は症例報告が散見される程度で,全体像は不明な点が多い.自験6例の経過から臨床像の把握と治療方針の検討を行った.基礎疾患はFallot四徴2例,完全型房室中隔欠損兼肺動脈閉鎖・純型肺動脈閉鎖・三尖弁閉鎖・総動脈幹遺残各1例であった.mBTS閉塞時に感染性閉塞と診断したのは2例のみで,残りは閉塞判明後の精査で感染性閉塞と診断した.起因菌はメチシリン耐性コアグラーゼ陰性ブドウ球菌3例,サルモネラ菌・セラチア菌・表皮ブドウ球菌各1例であった.3例でmBTS近位側吻合部に仮性瘤を形成した.新規の肺血流供給源をoriginal BTS(2例),心臓カテーテル治療によるmBTS再開通・右室流出路形成術・右室肺動脈導管サイズアップ(各1例)により確保した.右室肺動脈導管サイズアップ以外では肺血流確保が不可能な1例のみ人工物を使用した.心臓カテーテル治療でmBTSが再開通した1例以外で感染巣(人工血管グラフト,仮性瘤)を除去した.mBTS感染性閉塞では,閉塞との関連を疑う病歴の乏しい症例もあり注意を要する.mBTS近位側吻合部の仮性瘤は感染性閉塞を示唆する.(1)低酸素血症への対応,(2)新たな肺血流供給源の確立,(3)人工物を使用しない術式,(4)感染巣除去,を基本に症例ごとに治療方針を検討する必要がある.

Infective obstruction of a modified Blalock–Taussig shunt (mBTS) has remained poorly investigated. Here, we present six cases of this condition and discuss the therapeutic strategy. The underlying congenital heart diseases were tetralogy of Fallot (n=2), complete atrioventricular septal defect with pulmonary atresia (n=1), pulmonary atresia with an intact ventricular septum (n=1), tricuspid atresia (n=1), and truncus arteriosus communis persistens (n=1). Only two cases had infection during the diagnosis of mBTS obstruction. The bacterial pathogens were methicillin-resistant coagulase-negative staphylococci (n=3), Salmonella spp. (n=1), Serratia marcescens (n=1), and Staphylococcus epidermidis (n=1). Furthermore, three cases had pseudoaneurysm at the proximal anastomotic site of the mBTS. Pulmonary blood flow supply was established by mBTS recanalization through catheterization (n=1), right ventricular outflow tract reconstruction (n=1), original BTS utilization (n=2), and a larger-sized right ventricle-pulmonary artery conduit (n=1). Only one patient with no alternative therapeutic options was applied with prosthetic materials. Foci of infection, such as artificial shunt graft and pseudoaneurysm, were completely removed, except in one patient who underwent effective catheterization. In conclusion, possible infection must be considered in patients with mBTS obstruction. Pseudoaneurysm at the proximal anastomotic site of the mBTS suggests infective obstruction. Furthermore, the therapeutic strategy includes 1) treatment for hypoxemia, 2) establishment of a new supply of pulmonary blood flow, 3) surgery without using prosthetic materials, and 4) removal of infection foci.

Key words: Blalock–Taussig shunt; infective obstruction; pseudoaneurysm; infective endocarditis

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