An Anomalous Unilateral Single Pulmonary Vein Associated with a Bone Morphogenetic Protein Receptor II Gene Mutation

  • Koyama Kazuya
    Division of Respiratory Medicine, Toho University School of Medicine, Japan
  • Sano Go
    Division of Respiratory Medicine, Toho University School of Medicine, Japan
  • Hata Yoshinobu
    Division of Chest Surgery, Toho University School of Medicine, Japan
  • Shiraga Nobuyuki
    Department of Radiology, Toho University School of Medicine, Japan
  • Ota Hiroki
    Division of Respiratory Medicine, Toho University School of Medicine, Japan
  • Sugino Keishi
    Division of Respiratory Medicine, Toho University School of Medicine, Japan
  • Isobe Kazutoshi
    Division of Respiratory Medicine, Toho University School of Medicine, Japan
  • Sakamoto Susumu
    Division of Respiratory Medicine, Toho University School of Medicine, Japan
  • Takai Yujiro
    Division of Respiratory Medicine, Toho University School of Medicine, Japan
  • Koezuka Satoshi
    Division of Chest Surgery, Toho University School of Medicine, Japan
  • Makino Takashi
    Division of Chest Surgery, Toho University School of Medicine, Japan
  • Otsuka Hajime
    Division of Chest Surgery, Toho University School of Medicine, Japan
  • Sato Fumitomo
    Division of Chest Surgery, Toho University School of Medicine, Japan
  • Sasamoto Shuichi
    Division of Chest Surgery, Toho University School of Medicine, Japan
  • Iyoda Akira
    Division of Chest Surgery, Toho University School of Medicine, Japan
  • Kurosaki Atsuko
    Department of Diagnostic Radiology, Toranomon Hospital, Japan
  • Homma Sakae
    Division of Respiratory Medicine, Toho University School of Medicine, Japan

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抄録

Anomalous unilateral single pulmonary vein (AUSPV), a rare congenital anomaly, is associated with an aberrant course but normal drainage, and resembles arteriovenous malformation (AVM). We treated a 26-year-old man with AUSPV in the right lung and an anomalous segmental pulmonary vein in the left lung. CT revealed a tortuous vascular shadow with an enhancement pattern identical to that of the pulmonary vein, suggesting AUSPV. This was confirmed by pulmonary angiography. Although pulmonary AVMs were not detected on angiography, microvascular AVMs could not be excluded because delayed bubbles appeared on contrast echocardiography. A genetic examination revealed a missense mutation of BMPR2.<br>

収録刊行物

  • Internal Medicine

    Internal Medicine 53 (5), 461-466, 2014

    一般社団法人 日本内科学会

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