Griswold Catherine, Durica Allison R, Dennis Larry G, Jewell Ann F
Carilion Clinic Maternal-Fetal Medicine, Roanoke, Virginia USA; and Virginia Tech Carilion School of Medicine, Roanoke, Virginia, USA.
J Ultrasound Med. 2018 Apr;37(4):1033-1037. doi: 10.1002/jum.14428. Epub 2017 Sep 29.
There are multiple etiologies for fetal dilated bowel loops on ultrasonography (US), and we present a unique case of male siblings with a forkhead box P3 (FOXP3) mutation. Both children presented with fetal bowel anomalies on prenatal US. Family histories of cystic fibrosis and immune dysregulation, polyendocrinopathy, enteropathy, X-linked (IPEX) syndrome were reported. Amniocentesis in both pregnancies identified a normal male karyotype and the familial mutation associated with IPEX syndrome. IPEX syndrome is one of a group of conditions known as congenital diarrhea disorders. Other congenital diarrhea disorder cases have presented with similar prenatal US findings. As a result of these associations, we suggest considering IPEX syndrome as a potential cause of fetal bowel anomalies, particularly with a known family history. However, continued research into the phenotypic and genotypic correlations for IPEX syndrome is likely needed to better understand this possible prenatal presentation.
超声检查(US)发现胎儿肠袢扩张有多种病因,我们报告了一例独特的患有叉头框P3(FOXP3)突变的男性同胞病例。两个孩子在产前超声检查中均出现胎儿肠道异常。据报告有囊性纤维化家族史以及免疫失调、多内分泌腺病、肠病、X连锁(IPEX)综合征。两次妊娠的羊水穿刺均确定为正常男性核型以及与IPEX综合征相关的家族突变。IPEX综合征是一组被称为先天性腹泻疾病的病症之一。其他先天性腹泻疾病病例也有类似的产前超声检查结果。鉴于这些关联,我们建议将IPEX综合征视为胎儿肠道异常的潜在原因,尤其是在有已知家族史的情况下。然而,可能需要继续研究IPEX综合征的表型和基因型相关性,以便更好地理解这种可能的产前表现。