A case of branchial arch syndrome developing polyhydramnios occurring during the third trimester of pregnancy

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  • TSUKIOKA Miho
    Department of Obstetrics and Gynecology, Osaka City University Graduate School of Medicine
  • TACHIBANA Daisuke
    Department of Obstetrics and Gynecology, Osaka City University Graduate School of Medicine
  • YAMADA Shiori
    Department of Obstetrics and Gynecology, Kashiwara Municipal Hospital
  • NAKANO Akemi
    Department of Obstetrics and Gynecology, Osaka City University Graduate School of Medicine
  • TERADA Hiroyuki
    Department of Obstetrics and Gynecology, Osaka City University Graduate School of Medicine
  • SAITO Mika
    Department of Pediatrics, Osaka City University Graduate School of Medicine
  • KOYAMA Masayasu
    Department of Obstetrics and Gynecology, Osaka City University Graduate School of Medicine
  • ISHIKO Osamu
    Department of Obstetrics and Gynecology, Osaka City University Graduate School of Medicine

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Other Title
  • 妊娠後期に羊水過多を呈した鰓弓症候群の1例
  • 症例報告 妊娠後期に羊水過多を呈した鰓弓症候群の1例
  • ショウレイ ホウコク ニンシン コウキ ニ ヨウスイ カタ オ テイシタ シキュウ ショウコウグン ノ 1レイ

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Description

Branchial arch syndrome is a congenital anomaly that presents with hypoplasia of tissues derived from the first and second branchial arches and is characterized by microtia and mandibular hypoplasia. We describe a case of branchial arch syndrome diagnosed postnatally after detection of polyhydramnios during the third trimester of pregnancy. A 39-year-old primiparous woman with unremarkable history had been followed in our department since the first trimester of pregnancy. Polyhydramnios was detected, but there were not any anomalies suggesting gastrointestinal or other disorders of the fetus at 35 weeks of gestation. At 40 weeks and 0 days, the patient was admitted due to progressive edema of her leg. Amniotic fluid index (AFI) at the time of admission was 25.1 cm. At 40 weeks and four days, late decelerations of fetal heart rate were detected on cardiotocography (CTG), and emergency cesarean section was performed under general anesthesia due to non-reassuring fetal status. Female baby with a birth weight of 2602g was born (Apgar scores at 1 and 5 minutes were 1 and 6 points respectively; pH of umbilical arterial blood was 7.168; base excess was -9.6). The baby was intubated and supported by artificial respiration because of neonatal asphyxia. She exhibited bilateral macrostomia a high-arched palate, bilateral low-set ears, microgenia, and bilateral accessory auricles. These findings led to the diagnosis of the branchial arch syndrome. Respiratory status improved rapidly and she was extubated on the first day after birth. She had difficulties with swallowing milk due to bilateral macrostomia and therefore tube feeding was required until the third day after birth, followed by oral injection of milk with a syringe. At the age of three months surgical correction of the bilateral macrostomia and bilateral accessory auricles was performed. As of 16 months old, the child’s physical and mental development appeared favorable. It has been reported in the scientific literature that nearly 40% of cases of branchial arch syndrome show polyhydramnios due to fetal dysphagia. The present case suggests that the fetal face should be examined in details in consideration of the present syndrome when polyhydramnios of unknown origin is detected. [Adv Obstet Gynecol, 65(3) : 290-294, 2013 (H25.8)]

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