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 33(3): 215-220 (2017)
doi:10.9794/jspccs.33.215

症例報告Case Report

胎児鏡下胎盤吻合血管レーザー凝固術後も僧帽弁閉鎖不全が残存した双胎間輸血症候群受血児の1例The Case of a Recipient Twin Suffering from Twin-twin Transfusion Syndrome Whose Mitral Regurgitation Did Not Improve after Fetoscopic Laser Photocoagulation

1大垣市民病院小児循環器新生児科Department of Pediatric Cardiology and Neonatology, Ogaki Municipal Hospital ◇ Gifu, Japan

2福岡市立こども病院循環器科Fukuoka Children’s Hospital ◇ Fukuoka, Japan

3愛知県済生会リハビリテーション病院Aichi Saiseikai Rehabilitation Hospital ◇ Aichi, Japan

受付日:2016年11月25日Received: November 25, 2016
受理日:2017年3月13日Accepted: March 13, 2017
発行日:2017年5月1日Published: May 1, 2017
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双胎間輸血症候群(twin-twin transfusion syndrome; TTTS)の受血児における僧帽弁閉鎖不全(mitral regurgitation; MR)の頻度は6~15%とされ,予後不良因子の1つである.我々は心拡大,胎児水腫を認めたTTTSに対し胎児鏡下胎盤吻合血管レーザー凝固術(fetoscopic laser photocoagulation; FLP)を施行後も胎児期に重度の僧帽弁閉鎖不全は継続し,出生後に軽度へ改善した症例を経験したので報告する.症例はTTTSの受血児,在胎19週4日に胎児水腫のため転院し重度のMRを指摘された.在胎19週5日にFLPを施行し,徐々に羊水量の差と胎児水腫は改善した.在胎27週0日の胎児超音波検査では心胸郭断面積比47%,重度のMRを認めた.在胎32週4日に前期破水のため帝王切開で出生した.出生後の心臓超音波検査ではMRは重度で,僧帽弁に肥厚と前尖の軽度逸脱を認めた.日齢1に肺出血を生じたが,動脈管の閉鎖,呼吸状態や肺高血圧の改善に伴いMRは軽快した.僧帽弁は肥厚と前尖の逸脱を認め,容量負荷による機能性MRだけではないと考えられたが,1歳まで再増悪はなかった.

A recipient twin with twin-twin transfusion syndrome (TTTS) exhibited 6~15% mitral regurgitation (MR), and hence, a poor prognosis. Fetoscopic laser photo (FLP) coagulation was performed on the recipient twin who presented with cardiomegaly and hydrops fetalis, after which severe mitral and tricuspid regurgitation and cardiomegaly failed to resolve. Here, we present the case of a recipient twin with TTTS. The mother, at 19 weeks and 4 days of gestation, was referred for fetal hydrops. Ultrasonography indicated severe MR. FLP was performed at 19 weeks and 5 days, after which the hydrops fetalis resolved gradually. At 27 weeks, we noted cardiomegaly and severe MR in the recipient. The patient was delivered by an emergency caesarian section, which had to be performed because of premature rupture of the membranes at 32 weeks and 4 days. Postnatal cardiac ultrasonography indicated severe mitral regurgitation, with the anterior leaflet of the mitral valve prolapsed and thickened. Despite the presence of pulmonary hemorrhage at the age of 1 day, the patient was treated with indomethacin to close the ductus arteriosus, thereby improving MR and the respiratory state, while resolving pulmonary hypertension. We concluded that the cause of MR was volume overload in the left atrium and mild abnormalities in the mitral valve. MR did not worsen later during infancy.

Key words: fetal cardiology; twin-twin transfusion syndrome; recipient; mitral regurgitation; fetoscopic laser photocoagulation

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