Knowles S, Thomas R M, Lindenbaum R H, Keeling J W, Winter R M
Queen Victoria Hospital, South Australia.
Arch Dis Child. 1988 Jul;63(7 Spec No):723-6. doi: 10.1136/adc.63.7_spec_no.723.
Pulmonary agenesis is an uncommon anomaly that has been reported in isolation and in association with other congenital defects. Such defects include oesophageal atresia, cardiac malformation, horseshoe kidney, and anal atresia. Over a period of three years we have seen five neonates or fetuses with unilateral agenesis of the lung. All the cases had three or more anomalies seen in the VACTERL sequence in addition to the pulmonary atresia. None had a tracheo-oesophageal fistula. None were the products of consanguineous marriages. There had been no recurrence of this range of defects in any of the families at the time of writing. We suggest that pulmonary agenesis may occur as an alternative to tracheo-oesophageal fistula in the VACTERL sequence.
肺不发育是一种罕见的异常情况,曾有孤立报道,也有与其他先天性缺陷相关的报道。此类缺陷包括食管闭锁、心脏畸形、马蹄肾和肛门闭锁。在三年时间里,我们共诊治了5例单侧肺不发育的新生儿或胎儿。所有病例除肺不发育外,在VACTERL序列中还存在三种或更多异常。无一例有气管食管瘘。无一例为近亲结婚的产物。在撰写本文时,任何一个家庭中均未出现过此类缺陷的复发情况。我们认为,在VACTERL序列中,肺不发育可能是气管食管瘘的一种替代情况。