Department of Neurology, Children's National Hospital, Washington, District of Columbia.
Prenatal Pediatrics Institute, Children's National Hospital, Washington, District of Columbia; Department of Neurology, The George Washington University School of Medicine and Health Sciences, Washington, District of Columbia; Department of Pediatrics, The George Washington University School of Medicine and Health Sciences, Washington, District of Columbia.
Pediatr Neurol. 2022 Nov;136:8-14. doi: 10.1016/j.pediatrneurol.2022.07.011. Epub 2022 Aug 5.
Absent septum pellucidum (ASP) is a brain abnormality often associated with neuroanatomic abnormalities including septo-optic dysplasia (SOD). We aimed to determine how frequently prenatally diagnosed isolated ASP is confirmed by postnatal imaging and to examine clinical outcomes for ASP.
This was a retrospective study of maternal-fetal dyads referred to Children's National Hospital from January 1, 2012, to June 30, 2019. We included cases with fetal diagnosis of isolated or complex ASP. Diagnosis was based on ASP and the presence or absence of additional neuroanatomic findings. Data included obstetric and birth history, genetic testing, imaging, and neurodevelopmental outcomes.
ASP was diagnosed in 35 fetuses. Of 17 fetuses with isolated ASP, 10 had postnatal evaluation. In five (50%) isolated ASP cases, postnatal imaging revealed additional brain abnormalities. The five children with postnatally confirmed isolated ASP had lower rates of hydrocephalus (0% vs 54%) and abnormal feeding (0% vs 20%), hearing (0% vs 14%), and vision (0% vs 14%) than those with complex ASP (n = 17). Children with isolated ASP had lower rates of developmental delay (33% vs 50%) and seizures (11% vs 30%) than children with complex ASP. One child with prenatal isolated ASP was diagnosed with SOD (10%).
Few children with prenatally diagnosed isolated ASP had SOD diagnosed postnatally. Overall, children with isolated ASP demonstrate better outcomes than children with complex ASP. Fetal magnetic resonance imaging is a useful tool to evaluate the septum pellucidum and may reveal additional abnormalities that can impact prognosis and affect prenatal counseling.
透明隔缺如(ASP)是一种经常与神经解剖异常相关的脑异常,包括隔-视神经发育不良(SOD)。我们旨在确定产前诊断为孤立性 ASP 的病例在多大程度上可通过产后影像学检查得到证实,并研究 ASP 的临床结局。
这是一项回顾性研究,纳入了 2012 年 1 月 1 日至 2019 年 6 月 30 日期间转诊至儿童国家医院的母婴对子。我们纳入了病例 ASP 诊断为孤立性或复杂性。诊断基于 ASP 以及是否存在其他神经解剖学发现。数据包括产科和分娩史、基因检测、影像学和神经发育结局。
35 例胎儿被诊断为 ASP。17 例 ASP 中,有 10 例进行了产后评估。在 5 例(50%)孤立性 ASP 病例中,产后影像学检查显示存在其他脑异常。5 例经产后证实为孤立性 ASP 的患儿脑积水(0% vs 54%)、喂养异常(0% vs 20%)、听力(0% vs 14%)和视力(0% vs 14%)的发生率低于复杂性 ASP 患儿(n=17)。孤立性 ASP 患儿的发育迟缓(33% vs 50%)和癫痫(11% vs 30%)发生率低于复杂性 ASP 患儿。1 例产前孤立性 ASP 患儿被诊断为 SOD(10%)。
少数产前诊断为孤立性 ASP 的患儿在产后被诊断为 SOD。总体而言,孤立性 ASP 患儿的结局优于复杂性 ASP 患儿。胎儿磁共振成像(MRI)是评估透明隔的有用工具,可能会发现其他影响预后并影响产前咨询的异常。