Suppr超能文献

产前诊断为不对称性脑室扩大、半球间囊肿和胼胝体发育不全的婴幼儿的神经学和临床结局

Neurological and clinical outcomes in infants and children with a fetal diagnosis of asymmetric ventriculomegaly, interhemispheric cyst, and dysgenesis of the corpus callosum.

作者信息

Limoges Natalie, Ostrander Betsy, Kennedy Anne, Woodward Paula J, Bollo Robert J

机构信息

1Division of Neurological Surgery, University of Vermont Medical Center, Burlington, Vermont.

2Division of Pediatric Neurology, Department of Pediatrics, University of Utah, Primary Children's Hospital, Salt Lake City.

出版信息

J Neurosurg Pediatr. 2021 Nov 19;29(3):283-287. doi: 10.3171/2021.9.PEDS21252. Print 2022 Mar 1.

Abstract

OBJECTIVE

Advances in prenatal imaging have facilitated improvements in the fetal diagnosis of congenital anomalies. Asymmetric ventriculomegaly, interhemispheric cyst, and dysgenesis of the corpus callosum (AVID) is a constellation of congenital anomalies reported in fetal imaging. However, few data are available regarding postnatal outcomes of infants and children with a fetal diagnosis of AVID. The authors sought to report the neurodevelopmental outcomes of patients diagnosed with AVID before birth at a single institution.

METHODS

An institutional fetal imaging database was queried to identify cases with ventriculomegaly, interhemispheric cyst, and dysgenesis of the corpus callosum over a 10-year study period from 2000 to 2019. Overall, 41 maternal-infant dyads who met imaging criteria for AVID were identified; medical records were reviewed for prenatal variables including gestational age at birth, perinatal complications including fetal demise, and postnatal variables including demographics, mortality, hydrocephalus diagnosis and management, epilepsy, and neurodevelopmental outcomes at 2 years or the last follow-up.

RESULTS

Among 41 patients, 25 (61%) were male. A slight majority of patients (55%) were born before 36 weeks of gestational age, and 27 patients (68%) were delivered via cesarean section because their head size precluded vaginal delivery. There were 8 incidences of fetal demise, 1 pregnancy was terminated, and 32 patients were born alive. Neonatal or early infant death occurred in 5 patients. Two children died during follow-up after the neonatal period (ages 7 months and 7 years). Twenty-six children survived to at least the 2-year follow-up, all of whom required treatment for hydrocephalus. Of those 26 children, 12 (46%) had a diagnosis of epilepsy, 14 (54%) could sit independently, 4 (16%) were in mainstream school, 16 (62%) had expressive language, and 7 (28%) had near-normal development without seizures.

CONCLUSIONS

Among 41 maternal-fetal dyads with AVID, a majority of children survived to the 2-year follow-up, although all developed hydrocephalus. Many continued to have seizures, but expressive language use, attendance at mainstream school, and near-normal development without seizures were not infrequent. These data are critical for prenatal counseling and to establish the natural history of a diagnosis with limited outcome data.

摘要

目的

产前影像学的进展促进了先天性异常胎儿诊断的改善。不对称性脑室扩大、半球间囊肿和胼胝体发育不全(AVID)是胎儿影像学中报道的一组先天性异常。然而,关于产前诊断为AVID的婴幼儿产后结局的数据很少。作者试图报告在单一机构中产前诊断为AVID的患者的神经发育结局。

方法

查询机构胎儿影像数据库,以确定2000年至2019年10年研究期间患有脑室扩大、半球间囊肿和胼胝体发育不全的病例。总体而言,确定了41对符合AVID影像标准的母婴;查阅病历以获取产前变量,包括出生时的孕周、围产期并发症(包括胎儿死亡),以及产后变量,包括人口统计学、死亡率、脑积水的诊断和治疗、癫痫以及2岁或最后一次随访时的神经发育结局。

结果

41例患者中,25例(61%)为男性。大多数患者(55%)在孕36周前出生,27例患者(68%)因头部大小不适合阴道分娩而行剖宫产。有8例胎儿死亡,1例妊娠终止,32例患者存活。5例患者发生新生儿或早期婴儿死亡。2例儿童在新生儿期后的随访期间死亡(年龄分别为7个月和7岁)。26例儿童存活至至少2年随访,所有这些儿童均需要治疗脑积水。在这26例儿童中,12例(46%)诊断为癫痫,14例(54%)能独立坐立,4例(16%)在主流学校上学,16例(62%)有表达性语言能力,7例(28%)发育接近正常且无癫痫发作。

结论

在41对患有AVID的母婴中,大多数儿童存活至2年随访,尽管所有人都出现了脑积水。许多儿童继续有癫痫发作,但表达性语言使用、在主流学校上学以及发育接近正常且无癫痫发作的情况并不少见。这些数据对于产前咨询以及确定结局数据有限的诊断的自然病史至关重要。

文献AI研究员

20分钟写一篇综述,助力文献阅读效率提升50倍。

立即体验

用中文搜PubMed

大模型驱动的PubMed中文搜索引擎

马上搜索

文档翻译

学术文献翻译模型,支持多种主流文档格式。

立即体验