Wang H Z, Soong W J, Hwang B T
Pediatric Department, Veterans General Hospital, Taipei, R.O.C.
Zhonghua Yi Xue Za Zhi (Taipei). 1990 Oct;46(4):240-3.
Asphyxiating thoracic dystrophy (ATD) is a rare case of autosomal recessive disease. We report a case of full term female infant, who was noted to have small chest cage with severe respiratory distress soon after birth. On physical examination. The chest wall was fixed and small with the narrowest circumference about 29 cm in size, general cyanosis and distended abdomen were noted, there was a umbilical hernia around 1 cm in diameter. Otherwise, no other gross anomalies were found. Radiologic studies revealed short and horizontal ribs, small lung volume but depressed diaphragm, the clavicles and the spine were normal. The abdomen and long bone series all showed negative findings. The patient died of respiratory failure at 18 hours after birth. Autopsy was documented as a case of ATD. According to family history, there was one sibling die in the similar condition, although no autopsy available. Because the disease is transmitted as autosomal recessive trait, so 25% of next sibling will get the same condition. Therefore genetic consulting is necessary.
窒息性胸廓发育不良(ATD)是一种罕见的常染色体隐性疾病。我们报告一例足月女婴,出生后不久即被发现胸廓小并伴有严重呼吸窘迫。体格检查发现,胸壁固定且小,最窄处周长约29厘米,全身发绀,腹部膨隆,有一个直径约1厘米的脐疝。除此之外,未发现其他明显异常。影像学检查显示肋骨短且水平,肺容积小但膈肌低平,锁骨和脊柱正常。腹部和长骨系列检查均无异常发现。患儿出生后18小时死于呼吸衰竭。尸检证实为ATD病例。根据家族史,有一个兄弟姐妹死于类似情况,不过未进行尸检。由于该疾病以常染色体隐性性状遗传,所以下一胎有25%的概率会患同样的疾病。因此,进行遗传咨询是必要的。