A Case of an Anomalous Systemic Arterial Supply to Normal Basal Segments Treated by Transection of the Abnormal Vessel

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  • Matsuura Rei
    Department of Pediatric Surgery, Osaka University Graduate School of Medicine Department of Pediatric Surgery, Osaka Medical Center and Research Institute for Maternal and Child Health
  • Tazuke Yuko
    Department of Pediatric Surgery, Osaka Medical Center and Research Institute for Maternal and Child Health
  • Goda Taro
    Department of Pediatric Surgery, Izumiotsu Municipal Hospital
  • So Hideki
    Department of Pediatric Surgery, Osaka Medical Center and Research Institute for Maternal and Child Health
  • Ishii Tomohiro
    Department of Pediatric Surgery, Nara Hospital Kinki University Faculty of Medicine
  • Yamanaka Hiroaki
    Department of Pediatric Surgery, Osaka Medical Center and Research Institute for Maternal and Child Health
  • Nomura Motonari
    Department of Pediatric Surgery, Osaka Medical Center and Research Institute for Maternal and Child Health
  • Deguchi Koichi
    Department of Pediatric Surgery, Osaka Medical Center and Research Institute for Maternal and Child Health
  • Yoneda Akihiro
    Department of Pediatric Surgery, Osaka Medical Center and Research Institute for Maternal and Child Health

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Other Title
  • 異常血管の切離を施行した肺底動脈体動脈起始症の1例
  • 症例報告 異常血管の切離を施行した肺底動脈体動脈起始症の1例
  • ショウレイ ホウコク イジョウ ケッカン ノ キリハナシ オ シコウ シタ ハイテイ ドウミャクタイ ドウミャクキシショウ ノ 1レイ

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Abstract

The anomalous systemic arterial supply to the normal basal segment of the left lung, classically called Pryce’s type I intralobar pulmonary sequestration, rarely causes severe symptoms during early infancy. A male newborn with 21 trisomy developed tachypnea at 8 days of age and required management by nasal directional positive airway pressure (DPAP). A chest X-ray showed cardiomegaly, and echocardiography showed an atrial septum defect (ASD) accompanied by right heart failure. Contrast-enhanced computed tomography found an anomalous vessel arising from the descending thoracic aorta and returning to the left pulmonary vein through the left lower lung. These findings led to a diagnosis of anomalous systemic arterial supply to the normal basal segments (intralobar pulmonary sequestration Pryce type I). His pulmonary hypertension deteriorated owing to aortopulmonary shunting through the anomalous vessel and left-to-right intracardiac shunting through the ASD. The medical treatments for the right heart overload were ineffective and, as a result, nasal DPAP continued to be required. On postnatal day 43, the anomalous vessel was transected surgically, and a subsequent left lower lobectomy was avoided because the blood supply seemed to be maintained by the bronchial arteries. Soon after the surgery, the patient’s circulatory and respiratory systems were stabilized. The patient’s respiratory support was discontinued on postoperative day 3. The patient’s course remains good 7 months after surgery.

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