日本未熟児新生児学会雑誌　第17巻　第1号　47～50頁（2005年）

日本未熟児新生児学会雑誌　17(1)：47-50；2005　

受理日：平成16．07．02

未熟児動脈管開存症に対するインドメサシンとデキサメサゾンの併用療法が奏効したWilson-Mikity症候群の一例

A case of Wilson-Mikity syndrome with patent ductus arteriosus in which combined therapy with indomethacin and dexamethasone was effective

＊1東京医科大学　小児科，＊2東京女子医科大学日本心臓血圧研究所　循環器小児科

＊1Department of Pediatrics, Tokyo Medical University，＊2Pediatric Cardiology, The Heart Institute of Japan, Tokyo Women’s Medical University

佐藤　智＊1・高見　剛＊1＊2・金高由季＊1・斉藤哲也＊1・武井章人＊1・宮島　祐＊1・星加明徳＊1・門間和夫＊2

Satoshi SATO＊1，Takeshi TAKAMI＊1＊2，Yuki KANETAKA＊1，Tetuya SAITO＊1， Yukito TAKEI＊1，Tasuku MIYAJIMA＊1，Akinori HOSHIKA＊1，Kazuo MOMMA＊2

Key Words：PDA，indomethacin，dexamethasone，Wilson-Mikity syndrome

　症例は在胎27週2日，出生体重1,163gの女児。出生直後から人工呼吸器にて管理されていた。出生時IgM 59mg/dL，絨毛膜羊膜炎を認め，日齢3から胸部単純X線写真で泡沫状陰影が見られたためWilson-Mikity症候群と診断した。
　日齢20，感染を契機に動脈管の再開通を認めた。インドメサシン（IND）投与により一旦は閉鎖が認められたが，その後も再開通を繰り返した。日齢37からデキサメサゾン（DXA：0.3mg/kg/day，0.2mg/kg/day，0.1mg/kg/day，各3日間）とIND（0.2mg/kg/回×5回）の併用療法を開始し，日齢44に閉鎖を確認した。日齢45に抜管し，その後も動脈管の再開通は認めていない。
　INDとDXAの併用療法は相乗的に動脈管収縮をきたすと考えられ，特に急性期以降に認められた未熟児動脈管開存症の再発例では，治療の選択肢の一つになると考えられた。

　A female infant was born at 27 weeks and 2 days of gestation and her birth weight was 1163g. She received mechanical ventilation soon after birth. We suspected that she had Wilson-Mikity syndrome on the basis of the plasma immunoglobulin M（IgM）level（59mg/dL）upon admission, Chorioamnionitis, and diffuse and streaky infiltrate findings on chest X-ray from 3 days after birth.
　On day 20 of life, she showed infection signs and a patent ductus arteriosus（PDA）was diagnosed by cardiac ultrasound. The PDA was closed once by indomethacin therapy, but it re-opened later. Combined therapy withreoccurred with dexamethasone（0.3mg/kg/day, 0.2mg/kg/day, 0.1mg/kg/day； each for 3 days）and indomethacin（0.2mg/kg×5）was started from 37days of age. The ductus arteriosus was confirmed tobe closed at 44 days and she was successfully extubated the following day. The PDA did not re-open after that time.
　Combined therapy with indomethacin and dexamethasone may induce synergistic constriction of the ductus arteriosus. This method is an option for PDA treatment, especially in cases of repeated re-opening the PDA after the acute phase.