Usui Noriaki, Kamiyama Masafumi, Kimura Takuya, Kamata Shinkichi, Nose Keisuke, Fukuzawa Masahiro
Department of Pediatric Surgery, Osaka University Graduate School of Medicine, Osaka, Japan.
Pediatr Int. 2013 Feb;55(1):117-9. doi: 10.1111/j.1442-200X.2012.03620.x.
Although familial occurrence of congenital pyloric atresia (CPA) has been frequently reported in the past, many of these cases were associated with epidermolysis bullosa (EB), and familial isolated CPA was a relatively rare condition. We prenatally diagnosed and successfully treated a sibling of a subject with isolated CPA, who was diagnosed prenatally by fetal ultrasonography based on the findings of a distended stomach combined with polyhydramnios. The first case was a 2398-g female infant born at 36 weeks of gestation, who had been prenatally diagnosed as CPA. The second case, a younger sister of the first case, was a female infant weighing 2434 g, who had been also diagnosed as CPA by fetal ultrasonography at the check-up for the polyhydramnios of the same mother. Neither of the infants showed dermal lesions such as EB, and both underwent pyloroplasty with an excision of the pyloric membrane successfully after birth.
尽管过去曾频繁报道先天性幽门闭锁(CPA)的家族性发病情况,但其中许多病例与大疱性表皮松解症(EB)相关,而家族性孤立性CPA是一种相对罕见的病症。我们对一名孤立性CPA患者的同胞进行了产前诊断并成功治疗,该患者基于胃扩张合并羊水过多的超声检查结果在产前被诊断出来。第一例是一名妊娠36周出生的2398克女婴,产前被诊断为CPA。第二例是第一例的妹妹,是一名体重2434克的女婴,在其母亲因羊水过多进行检查时,通过胎儿超声检查也被诊断为CPA。两名婴儿均未出现如EB等皮肤病变,出生后均成功接受了幽门成形术并切除了幽门膜。