1 千葉県こども病院循環器科Division of Cardiology, Chiba Children’s Hospital ◇ Chiba, Japan
2 船橋市立医療センター小児科Division of Pediatrics, Funabashi Municipal Medical Center ◇ Chiba, Japan
先天性肺静脈狭窄ならびに閉鎖症は極めてまれな疾患である．学校検診での胸部単純写真の異常陰影を主訴に来院し，心臓カテーテル検査をはじめとする各検査によって先天性肺静脈狭窄症ならびに閉鎖症の診断に至った1例を経験した．右上肺静脈は左房へ開口せず，拡張し蛇行しながら右下肺静脈と合流し，anomalous unilateral single pulmonary vein（AUSPV）が認められた．先天性肺静脈狭窄症とAUSPVを合併した報告は今までなく本症例が初報告となる．左右肺血流不均衡は認めるものの，肺高血圧の合併はなく無治療で経過観察を行っている．
Congenital unilateral pulmonary vein stenosis is an extremely rare condition. Herein, we present a case of congenital unilateral pulmonary stenosis and atresia that we ultimately diagnosed by cardiac catheterization. The patient presented to the hospital with an abnormal shadow on a chest X-ray during a school examination. An aberrantly draining pulmonary vein developed from the atresia, suggesting an anomalous unilateral single pulmonary vein (AUSPV). This is the first report of a patient with congenital pulmonary stenosis with AUSPV. As there was no pulmonary hypertension at diagnosis, the patient was followed up without treatment.
Key words: congenital pulmonary stenosis; anomalous unilateral single pulmonary vein
© 2020 特定非営利活動法人日本小児循環器学会© 2020 Japanese Society of Pediatric Cardiology and Cardiac Surgery
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